Unusual Sequelae Research Articles

Insights into acromioclavicular joint ganglionic cyst: diagnosis and management

Acromioclavicular joint (ACJ) cysts are an uncommon and unusual sequela associated with shoulder pathophysiology1.It is a rare clinical entity that can affect patients’ cosmetic appearance as well as daily living activities. Limited number of case reports have been published till date in literature. This case report presents a rare example of a ganglion cyst taking origin from acromio clavicular joint in a 62-year-old female patient, outlining the clinical presentation, diagnostic approach, and successful surgical management.

933 Inguinal Bladder Hernia: Case Report and Literature Review

Abstract Background Partial bladder herniation into the inguinal canal has an incidence of 1-3%. Most patients are asymptomatic, and diagnosis is made incidentally upon imaging or during surgical repairs. Complete herniation of the bladder and distal ureters is rare, and few cases of resultant acute kidney injury or hyperkalaemia have been reported. This case presentation includes preoperative and intra-operative images, and literature review. Case presentation A 73-year-old gentleman with longstanding bilateral inguinal hernia, benign prostatic hyperplasia, type 2 diabetes mellitus, and obesity presented with new-onset lower urinary tract symptoms and intermittent pain over his right inguinal hernia. His PSA (Prostate-Specific Antigen) was raised, and imaging identified bilateral hydronephrosis, and right inguinal hernia containing distal ureters and bladder. He was added to the waiting list for elective hernia repair. Whilst awaiting repair, he was admitted with acute kidney injury and hyperkalaemia, which was managed with cardiac protection, urethral catheterisation, hydration, and serial blood tests. After stabilization, he underwent an open right inguinal hernia repair with mesh. His acute kidney injury resolved; however he failed trial without catheter (TWOC), and was discharged with a catheter pending TWOC clinic review. General Surgical Follow up is planned to discuss left inguinal hernia repair. Conclusion Awareness of bladder herniation into the inguinal canal is essential to avoid iatrogenic injury. Careful pre-operative planning and correction of physiological abnormalities is vital to ensure good surgical outcome. More unusual sequelae of untreated hernias are likely to become more frequent, if fewer elective hernias are performed.

ORGANIZING PNEUMONIA: AN UNUSUAL SEQUELA OF COVID-19 INFECTION.

COVID-19 pneumonia is one of the secondary causes of organizing pneumonia (OP).Early initiation of steroids in OP is associated with improvement in symptoms and prognosis.A prolonged course of steroids may be needed in COVID-induced OP.

New Presentation of CD27 Deficiency; Coronary Ectasia and COVID-19.

CD27 is a costimulatory receptor involved in the maturation of the innate and adaptive immunity. CD27, through interaction with CD70, plays a role in the control of Epstein-Barr virus (EBV) infection. CD27 deficiency leads to an immune dysregulation disease characterized by EBV susceptibility. Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) might put patients with primary immunodeficiency at risk for adverse outcomes. Chromogenic in situ hybridization (CISH) study was performed to detect EBV in the lymphoma tissue. Genetic analysis of the patient was done with Whole Exome Sequencing and detected variant was confirmed with PCR-Sanger sequencing. Here we report a 20-month-old boy with CD27 deficiency who developed lymphoma and coronary artery ectasia and had been infected with SARS-CoV-2. Clinical and laboratory findings were incompatible with atypical Kawasaki syndrome or multisystem inflammatory syndrome in children (MIS-C). As CD27 deficiency is a rare immune defect, publishing clinical data about the identified patient(s) can shed light on our knowledge about the related phenotype and the spectrum of clinical manifestations associated with CD27 deficiency. Thus, our findings expanded the spectrum of manifestations beyond EBV infection, highlighting this unusual cardiac sequela that could be related to EBV infection, lymphoma, or an underlying disease.

Diffuse Viral Myositis with Late Progression to Bilateral Avascular Necrosis of Femoral Head

Viral myalgia in post-COVID-19 has been a common presentation. Musculoskeletal affection with diffuse viral myositis (VM) has been an uncommon occurrence. The progression of myositis to avascular necrosis (AVN) of bilateral femoral heads has been rarely reported. The case study in a 60-year-old male presented with painful bilateral hips with difficulty in ambulation simulating pseudoparalysis. The magnetic resonance imaging confirmed diffuse myositis along both the hips which needed close observation. The serial imaging at 3-month intervals showed resolution of VM, however, progression to AVN of both femoral heads. The unusual affection was initially managed medically, however, later it necessitated a bilateral total hip replacement. An astute clinician's perspective during the radiological evaluation allowed to identify the progression. The long COVID-19 may present a debilitating musculoskeletal affection of myositis around hips with progression to AVN of femoral heads. The VM of severe acute respiratory syndrome coronavirus 2 etiology may have an unrelenting long-duration affection. A multidisciplinary team approach may help in overcoming the crisis.

An Unusual Sequela of Longstanding Gallstone Disease

An Unusual Sequela of Longstanding Gallstone Disease

Scalp osteomyelitis of the occipital bone in a neonate: An unusual sequel of a scalpel injury at birth

The Journal is the primary organ of Continuing Paediatric Medical Education in Sri Lanka. The journal also has a website. Free full text access is available for all readers.The Sri Lanka Journal of Child Health is now indexed in SciVerse Scopus (Source Record ID 19900193609), Index Medicus for South-East Asia Region (IMSEAR), CABI (Centre for Agriculture and Bioscience International Global Health Database), DOAJ and is available in Google, as well as Google Scholar.The policies of the journal are modelled on the Committee on Publication Ethics (COPE) Guidelines on Principles of Transparency and Best Practice in Scholarly Publishing. Sri Lanka Journal of Child Health is recognised by the International Committee of Medical Journal Editors (ICMJE) as a publication following the ICMJE Recommendations.

S1892 A Double Take on a 20-Year Double J-Stent

Introduction: The double J biliary stent is commonly used to relieve obstructions and aid in draining of pancreatic fluid. Indwelling or forgotten stents are rarely seen and their sequelae are unknown. We present the case of a patient who was lost to follow-up over twenty years ago following cholecystectomy and placement of double J stent. A routine esophagogastroduodenoscopy (EGD) for variceal surveillance was performed and discovered an ampullary fungating mass. Case Description/Methods: A 79 y/o female with a history of hypertension presented to the ED with worsening abdominal distention. She was a poor historian but denied alcohol use, drug use, or any history of liver disease. Her workup included an abdominal paracentesis removing greater than 7 liters of fluid with a SAAG indicating portal hypertension. She had abnormal liver function tests (LFTs) and CT imaging was consistent with cirrhotic morphology of the liver and a double-J stent present in CBD (Figure A). An EGD for further evaluation and for variceal vein monitoring was done revealing grade 2 varices but more significantly, at the site of ampulla of vater a large fungating mass was noted. The mass was medium-sized and polypoid with no bleeding. The CBD stent was in place with noted hyperproliferation of the tissue of the ampulla vs a periampullar duodenal mass (Figure B). A repeat EDG with EUS was performed, finding the hypoechoic non-circumferential mass endosonographically within the ampulla. The mass measured 24 mm by 14 mm in maximal cross-sectional diameter (Figure C). The lesion extended from the mucosa to the muscularis mucosa. The endosonographic borders were well-defined. There was sonographic evidence suggesting invasion into the deep mucosa (Layer 2). Filamentous microorganisms were identified on HE and GMS stain. No individual atypical AE1/AE3 positive epithelial cells were identified. Discussion: This case highlights the unusual sequelae of a retained double-J stent in a patient who was lost to follow up. It’s unknown why the stent was placed, likely during an intraoperative cholangiogram, and the patient did not recall her follow-up instructions. Several questions arise surrounding the mass such as, did it have malignant potential and what role, if any, did the stent have in the formation of the mass? Did colonization represent an increased risk of ascending cholangitis? The stent was left in at the conclusion of the case and the patient was extensively educated about follow-up and maintenance endoscopy.Figure 1.: CT evidence of pneumobilia, ampullary fungating mass, and EUS imaging.

Rare and Unusual Follow-up Sequelae of Coronavirus Disease 2019: Splenic Mucormycosis in a Renal Transplant Recipient

BackgroundCoronavirus disease 2019 (COVID-19) is associated with adverse outcomes in transplantation communities. Mucormycosis, although a rare infection, has been classically linked to organ transplantation and is associated with exceptionally high morbidity and mortality rates. In this pandemic era, the double infection of mucormycosis and COVID-19 is a lethal combination but is rarely described in the literature on organ transplantation.Case presentationThis article presents the case of a young kidney transplant recipient with diabetes who acquired severe COVID-19, followed by disseminated mucormycosis. The patient was a health care worker who developed severe COVID-19, for which he received remdesivir, anticoagulation, and dexamethasone. No immunomodulatory therapy was used. His maximum oxygen support was bilevel positive airway pressure ventilation. His sugar levels were frequently deranged during the stay. He developed secondary sepsis with Klebsiella, followed by nonhealing lung consolidation. He later developed pleural effusion and splenic abscess, which was detected incidentally. He underwent an emergency splenectomy, the culture of which yielded mucormycosis. Liposomal amphotericin B 5 mg/kg was administered. The patient deteriorated, and a repeat laparotomy yielded gastric perforation, with pus culture showing mucormycosis. The patient died after a long hospital stay.ConclusionsThe diagnosis and management of this dual infection during the pandemic is extremely challenging. In this case, the unusual location of mucormycosis complicating COVID-19 calls for a meticulous approach to opportunistic fungal infections in organ transplant recipients who are positive for COVID-19, especially in those patients with diabetes.

Long-term Risks of Persistent Ventral Spinal CSF Leaks in SIH

Superficial siderosis, bibrachial amyotrophy, and spinal cord herniation are unusual but serious long-term sequelae of persistent spontaneous spinal CSF leaks in patients with spontaneous intracranial hypotension (SIH), particularly ventral spinal CSF leaks. However, the risk of developing such sequelae has not been established in this population. We undertook this study to determine the risk of these serious complications of persistent ventral spinal CSF leaks. This cohort study was conducted using data from a prospectively maintained database of patients who meet the modified International Classification of Headache Disorders, 3rd edition criteria for SIH. The patient population consisted of a consecutive group of patients with SIH and persistent ventral spinal CSF leaks who were first seen within 1 year of onset of SIH symptoms and who had at least 1 year of follow-up. Among 51 patients with SIH and a persistent ventral spinal CSF leak, superficial siderosis developed in 6 patients and bibrachial amyotrophy in 2 patients during 280 patient-years of follow-up. The probability of these complications increased from 0% at 48 months to 4.5% (95% confidence interval [CI] 1.0%-28.0%) at 56 months, 10.5% (95% CI 3.0%-36.4%) at 96 months, 32.7% (95% CI 15.0%-62.8%) at 144 months, and 57.9% (95% CI 30.2%-87.6%) at 192 months. None of the patients developed spinal cord herniation. Among patients with SIH and a persistent ventral spinal CSF leak, the risk of developing serious long-term sequelae is considerable. This study shows that early treatment of a ventral spinal CSF leak offers a unique opportunity to prevent neurologic disability from superficial siderosis and bibrachial amyotrophy.

Heparin Skin Necrosis in Heparin-Induced Thrombocytopenia

A 66-year-old woman presented with painful abdominal skin necrosis where she had been injecting subcutaneous enoxaparin 1 week following elective knee arthroplasty (Figure). Her complete blood cell count and basic metabolic panel yielded unremarkable findings. Enoxaparin was discontinued, and a direct thrombin inhibitor was started. Heparin platelet factor 4 IgG antibody was strongly positive (2.065 OD), as was the serotonin release assay—findings consistent with the diagnosis of heparin skin necrosis and heparin-induced thrombocytopenia.1Warkentin T.E. Roberts R.S. Hirsh J. Kelton J.G. Heparin-induced skin lesions and other unusual sequelae of the heparin-induced thrombocytopenia syndrome: a nested cohort study.CHEST. 2005; 127: 1857-1861Abstract Full Text Full Text PDF PubMed Scopus (115) Google Scholar Ultrasound imaging was negative for deep venous thrombosis. The patient was discharged on a regimen of therapeutic rivaroxaban and did well without need for surgical debridement. Heparin skin necrosis is a rare presentation of heparin-induced thrombocytopenia that can complicate either low-molecular-weight or unfractionated heparin therapy. Normal platelet counts are common and do not preclude the diagnosis. A 66-year-old woman presented with painful abdominal skin necrosis where she had been injecting subcutaneous enoxaparin 1 week following elective knee arthroplasty (Figure). Her complete blood cell count and basic metabolic panel yielded unremarkable findings. Enoxaparin was discontinued, and a direct thrombin inhibitor was started. Heparin platelet factor 4 IgG antibody was strongly positive (2.065 OD), as was the serotonin release assay—findings consistent with the diagnosis of heparin skin necrosis and heparin-induced thrombocytopenia.1Warkentin T.E. Roberts R.S. Hirsh J. Kelton J.G. Heparin-induced skin lesions and other unusual sequelae of the heparin-induced thrombocytopenia syndrome: a nested cohort study.CHEST. 2005; 127: 1857-1861Abstract Full Text Full Text PDF PubMed Scopus (115) Google Scholar Ultrasound imaging was negative for deep venous thrombosis. The patient was discharged on a regimen of therapeutic rivaroxaban and did well without need for surgical debridement. Heparin skin necrosis is a rare presentation of heparin-induced thrombocytopenia that can complicate either low-molecular-weight or unfractionated heparin therapy. Normal platelet counts are common and do not preclude the diagnosis.

Bilateral subdural hygromas after endovascular coiling for ruptured aneurysmal subarachnoid hemorrhage: an unusual and rare complication

ABSTRACTSubarachnoid hemorrhage (SAH) with subdural hygroma (SH) was rarely reported after endovascular coiling. A 60-year-old male presented with impaired consciousness and convulsions due to SAH from a ruptured aneurysm. It was managed by endovascular coiling 20 h after the onset of symptoms. Serial brain imaging for 2 weeks revealed progressive bilateral SHs, more on contralateral side of leaking aneurysm. Management of SH was discussed in a multidisciplinary setting to be conservative as there was neither significant mass effect nor hydrocephalus. The patient recovered neurologically except for mild dysarthria. The SH persisted for 2 months and then cleared gradually. We concluded that SH may arise and become symptomatic as an unusual sequela of post-coiling of a ruptured intracranial aneurysm, in which the SH can complicate the clinical course of SAH. However, the symptomatic SH may resolve spontaneously and completely without any intervention, but needs meticulous neurological assessment and follow-up.

'Heart of stone': an unusual post-actinic sequela.

'Heart of stone': an unusual post-actinic sequela.

Unusual Presentation of COVID 19 - A Case report

An acute respiratory disease caused by a new strand of coronavirus (SARS-CoV-2), which was ûrstidentiûed in Wuhan, Hubei Province, China, quickly developed into a global pandemic. Commonpresentation associated with corona-virus disease 2019 (COVID-19) include fever, cough, and lesscommonly diarrhea, sore throat, chest pain, nausea, and vomiting , loss or attenuation of smell andtaste sensation and palpitations. Till now observed sequelae are ARDS, multi organ dysfunction,thrombotic events and coagulation disorder, myocarditis .With time clinicians are observing emergenceof new and unusual presentation, complications and sequelae of the disease. Our case report is of amiddle aged man with typical symptoms of COVID 19 who ultimately developed right sided tensionpneumothorax. Along with O2 therapy, anticoagulant, anti inflammatory drugs and other supportivetreatment he was managed with water seal drainage. Though till date COVID 19 is not very wellknown to us but development of tension Pneumothorax as asequelae is atypical as far observedwhich made this case interesting.
 Bangladesh J Medicine July 2020; 31(2) :102-106

Molecules in pathogenesis: angiotensin converting enzyme 2 (ACE2)

The renin–angiotensin system is mainly associated with the regulation of blood pressure, but recently many other functions of this system have been described. ACE2, an 805-amino acid monocarboxypeptidase type I...

Cutting Edge Courtship in Eighteenth-Century London

In 1759, an anonymous twenty-two year-old woman placed an advertisement for a husband in the London Daily Advertiser that inspired an unusual sequel: a pamphlet purporting to collect her responses. Exploring the context of this woman's actions and the letters themselves reveals attitudes regarding matrimony and the dangers of virtual social networks that feel surprisingly contemporary.

Acetazolamide-Triggered Acute Flaccid Paralysis and Toxic-Metabolic Encephalopathy in a Toddler with Gastroenteritis: A Case Report

AbstractAcetazolamide is an infrequently prescribed medication in the outpatient pediatric setting, as one of its major indications is for the treatment of glaucoma, which is a largely adult disease. Though incredibly rare, serious neurologic side effects such as stroke, flaccid paralysis, and coma can occur after its administration. This case presentation of acute flaccid paralysis with metabolic derangement underscores the unusual, yet possible, toxic-metabolic and neurologic sequelae that can occur in an already acidotic host (in this case, caused by gastroenteritis) with acetazolamide ingestion. Life-threatening conditions must always be ruled out in patients who present with encephalopathy, but a medication history was crucial in clinching this case's unifying diagnosis. While there are case reports highlighting the rare central nervous system toxicities associated with acetazolamide ingestion, to our knowledge, none exist in the pediatric literature.

SURG-08. GASTROINTESTINAL STROMAL TUMOR WITH INTRACRANIAL METASTASIS: CASE REPORT AND SYSTEMATIC REVIEW OF LITERATURE

BACKGROUND: Intracranial metastasis of Gastrointestinal Stromal Tumors (GIST) is rare but presents unique treatment challenges. We present a case of intracranial metastasis of GIST with a systematic review of the literature regarding this rare clinical scenario. METHODS: A systematic review of the literature was performed to identify cases of intradural GIST metastases to the brain. Additionally, a patient case of GIST is discussed. RESULTS: Out of the 18 articles included for analysis in this review and our present case, fifteen of nineteen patients were male, and mean age was 58 years old (range 15–80 years, median 60 years). The primary site of the GIST along with site of intraperitoneal metastasis was variable. There was a large predilection for brain metastasis to the cerebrum with only one to infratentorial elements. The tumors in seven of the cases involved the dura, and there was one case with metastasis to the pituitary. Eight patients died following treatment of their intracranial disease. CONCLUSIONS: Surgery remains the mainstay of intracranial metastatic GIST, however there are many reports of good responses to radiation or chemotherapy alone. More investigation is required to determine the best course of treatment for patients with this unusual sequela of GIST.

Severe dermatomyositis presenting as quadriparesis: An unusual sequel in elderly

Dermatomyositis, an idiopathic inflammatory myopathy, is one of the rare causes of limbs paresis. The characteristic feature is skin involvement which differentiates this from other inflammatory myopathy. Here we report a case of severe dermatomyositis as his Creatine Phosphokinase (CPK) level was 32,000 who presented with weakness in all four limbs without any dermatological signs. Keywords: Dermatomyositis, Creatinine kinase, Quadriparesis, Elderly.

The right eye abducens nerve palsy as a cranial neuropathy of dengue fever: The benefit of corticosteroids in an unusual dengue sequela

Dengue fever is very common in tropical climate countries and the number of reported cases in Malaysia shows an increasing trend recently, according to the Malaysian Clinical Practice Guidelines. Although dengue fever is common, cranial nerve mononeuropathy is a very rare manifestation in relation to other neurological-associated syndromes. We report a rare case of cranial mononeuropathy of dengue fever in Malaysia and highlight the option of steroid usage as an alternative treatment to hasten the neurological recovery. The patient, a 25-year-old healthy policeman, presented with symptomatic viral fever, which was serologically confirmed as dengue fever. He developed acute-onset binocular diplopia, which was secondary to right eye isolated abducens nerve palsy during the critical phase of dengue fever. His visual acuity was 6/6 in both eyes with slightly restricted abduction of the right eye, consistent with right abducens nerve palsy, which was confirmed with a Hess test. There was corresponding diplopia over the right paracentral visual field. Urgent contrasted brain imaging was done, which ruled out the life-threatening intracranial pathology; therefore, a diagnosis of possible subclinical inflammatory changes causing sixth nerve palsy was made. Subsequently, he was treated with intravenous methylprednisolone 500 mg daily for 3 days and regained full extraocular muscle movement after 1 week. Oral steroid was not initiated. In conclusion, although the isolated unilateral cranial mononeuropathy may improve spontaneously within a certain period of time, a short course of systemic corticosteroids may be considered to hasten the recovery, as it has a favourable outcome.