Chronic Lipoid Pneumonia in a 9-Year-Old Child Revealed by Recurrent Chest Pain

Lipoid pneumonia in children is a rare disorder due to accumulation of fatty oily material in the alveoli and usually associated with an underlying condition. In absence of obvious context, diagnosis remains difficult with nonspecific clinical and radiological features. We report the first case of voluntary chronic aspiration of olive oil responsible for exogenous lipoid pneumonia, in a previously healthy 9-year-old boy. Clinical presentation was atypical; LP was revealed by isolated chest pain. We discuss radiological and bronchial alveolar lavage characteristics suggestive of lipoid pneumonia.Conclusion. Lipoid pneumonia is a disease to be reminded of in children, which can occur with original findings in terms of etiology and clinical presentation.

Download Full-text

Painful, reappearing eruption in a medically complex 4-year-old

BMJ Case Reports ◽

10.1136/bcr-2020-239310 ◽

2021 ◽

Vol 14 (2) ◽

pp. e239310

Author(s):

Meredith Sooy ◽

Rachel L Randell ◽

Dmitry Tchapyjnikov ◽

Klaus Werner ◽

Kristina Nazareth-Pidgeon

Keyword(s):

Skin Biopsy ◽

Growth Retardation ◽

Clinical Presentation ◽

Heart Defects ◽

Pathogenic Mutation ◽

Rare Disorder ◽

Frequent Episodes ◽

First Case

A 4-year-old boy with atypical, complete DiGeorge and CHARGE (coloboma, heart defects, atresia choanae, growth retardation, genital abnormalities and ear abnormalities) syndromes presented with frequent episodes of a painful, markedly erythematous eruption associated with swelling. Evaluation revealed non-specific findings on skin biopsy at the time of eruption and no pathogenic mutation in the SCN9A gene. The patient was diagnosed with secondary erythromelalgia based on clinical presentation. Erythromelalgia is a rare disorder characterised by recurrent episodes of pain and erythema typically affecting the distal extremities. This case represents the first case of erythromelalgia in the setting of DiGeorge and CHARGE syndromes.

Download Full-text

Imaging of a rare disorder: macrodystrophia lipomatosa

South African Journal of Radiology ◽

10.4102/sajr.v14i2.425 ◽

2010 ◽

Vol 14 (2) ◽

pp. 39 ◽

Cited By ~ 1

Author(s):

Farhana Ebrahim Suleman ◽

Margaret Kisansa

Keyword(s):

The Other ◽

Rare Disorder ◽

Macrodystrophia Lipomatosa ◽

Radiological Features ◽

Extensive Search ◽

Full Expression ◽

First Case ◽

Congenital Condition ◽

History Of

Macrodystrophia lipomatosa (MDL) is described as a rare, non-hereditary, congenital condition presenting with localized macrodactyly and a proliferation of mesenchymal elements. There is in particular a marked increase in fibroadipose tissue(1). We describe two cases presenting to our department in a six month period, with a history of disproportionately large limbs since birth. While our first case demonstrated all the typical features of MDL, our second case failed to demonstrate osseous gigantism although the other features of MDL were present. An extensive search of the literature failed to yield any cases described without osseus gigantism but at the same time the other radiological features failed to fit in with any other related syndromes and the most appropriate diagnosis appears to be MDL. Thus we concluded that this may be a case of a rare, atypical MDL that was arrested or frustrated and therefore failed to demonstrate full expression of the syndrome.

Download Full-text

Hajdu-Cheney Syndrome: First Case Report from Bangladesh

Bangladesh Journal of Child Health ◽

10.3329/bjch.v36i2.13086 ◽

2012 ◽

Vol 36 (2) ◽

pp. 98-100

Author(s):

Shahana A Rahman ◽

Shohela Akhter ◽

Farhana Bayes ◽

Mashura Musharraf

Keyword(s):

Case Report ◽

Bone Resorption ◽

Child Health ◽

Connective Tissue ◽

Clinical Presentation ◽

Genetic Disorder ◽

Radiological Features ◽

First Case ◽

Wide Range ◽

Medical University

Hajdu-Cheney syndrome is an extremely rare genetic disorder of the connective tissue characterized by severe and excessive bone resorption leading to osteoporosis and a wide range of other potential symtromes. The clinical presentation and typical radiological features help in confirming the diagnosis. Hajdu-Cheney syndrome had not been reported previously in our country. Because of the rarity of the problem, we are interested to report a case of Hajdu-Cheney syndrome who was admitted in Bangabandhu Sheikh Mujib Medical University, Dhaka, Bangladesh. DOI: http://dx.doi.org/10.3329/bjch.v36i2.13086 Bangladesh J Child Health 2012; Vol 36 (2): 98-100

Download Full-text

Real world cost and health outcomes of patients presented with chest pain in England: which is the most cost-effective first-line test?

European Heart Journal ◽

10.1093/ehjci/ehaa946.3547 ◽

2020 ◽

Vol 41 (Supplement_2) ◽

Author(s):

A Tsiachristas ◽

H West ◽

E.K Oikonomou ◽

B Mihaylova ◽

N Sabharwall ◽

...

Keyword(s):

Chest Pain ◽

Health Outcomes ◽

Diagnostic Tests ◽

Clinical Presentation ◽

Cost Effective ◽

Funding Source ◽

First Line ◽

Nice Guidelines ◽

Exercise Ecg ◽

Stable Chest Pain

Abstract Background The National Institute for Health and Care Excellence (NICE) updated their guidance for the management of patients with stable chest pain and recommended that all patients undergo computed tomography coronary angiography (CTCA). This update has sparked a great deal of debate, and was followed by upgrade of CTCA into a Class I indication in the recent ESC guidelines. The cost-effectiveness of using CTCA as first line investigation is still unclear. Purpose To describe the current clinical pathway of patients with stable chest pain presented to outpatient clinics, assess the compliance with the updated NICE guideline, and explore the costs and health outcomes of different non-invasive diagnostic tests in real-world clinical setting. Methods We used data of 4,297 patients who attended chest pain clinics in Oxford between 1 January 2014 and 31 July 2018. Data included clinical presentation (e.g. age and previous cardiovascular conditions), diagnostic tests, outpatient visits, hospitalization, and hospital mortality and was compared between 6 alternative first-line diagnostic tests. Multinomial regressions were performed to estimate the probability of receiving each alternative and the associated cost after adjusting for clinical presentation. A decision tree was developed to describe the clinical pathway for each alternative first-line diagnostic in terms of subsequent diagnostic tests and treatments and to estimate the associated costs and life days. Results The proportion of patients who received CTCA as first line diagnostic test increased from 1% in 2014 to 17% in 2018, while the publication of the updated NICE guidelines in 2016 led to a threefold increase in this proportion. CTCA is less likely to be provided as a first-line diagnostic to patients who are younger age, males, smokers, and have angina, PVD, or diabetes. The standardised rate of hospital admission was the lowest in the exercise ECG cohort (0.35 admissions per 1,000 life-days) followed by the CTCA cohort (0.40 admissions per 1,000 life-days) while the latter cohort had the lowest standardised rate of cardiovascular treatment (2.74% per 1,000 life days). Stress echocardiography and MPS were associated with higher costs compared with CTCA, other ECG, and exercise ECG after adjusting for clinical presentation and days of follow-up. CTCA is the pathway most likely to be cost-effective, even compared to exercise ECG, while the other diagnostic alternatives are dominated (i.e. they cost more for less life-days). Conclusions Currently, the updated NICE guidelines for stable chest pain are implemented only to a fifth of the cases in England. Our findings support existing evidence that CTCA is the most-cost effective first-line diagnostic test for this population. Hopefully, this will inform the debate around the implementation of the guidelines and help commissioning and clinical decision processes worldwide. Funding Acknowledgement Type of funding source: Public grant(s) – National budget only. Main funding source(s): National Institute of Health Research Oxford Biomedical Research Centre

Download Full-text

Inflammatory Myofibroblastic Tumor of the Bladder: 2 Rare Cases Managed with Laparoscopic Partial Cystectomy

Case Reports in Urology ◽

10.1155/2016/4976150 ◽

2016 ◽

Vol 2016 ◽

pp. 1-3 ◽

Cited By ~ 1

Author(s):

Sofia Santos Lopes ◽

Andrea Furtado ◽

Rita Oliveira ◽

Ana Cebola ◽

Bruno Graça ◽

...

Keyword(s):

Inflammatory Myofibroblastic Tumor ◽

Clinical Presentation ◽

Treatment Approach ◽

Pathological Condition ◽

Partial Cystectomy ◽

Pathological Features ◽

Macroscopic Hematuria ◽

Voiding Symptoms ◽

First Case

Two cases of inflammatory myofibroblastic tumor (IMT) of the bladder are reported here. Both patients were male and presented with macroscopic hematuria; in the first case terminal hematuria was associated with irritative voiding symptoms. The second case was a smoker with hematuria unresponsive to medical treatment and anemia. Clinical presentation, pathological features, treatment, and prognosis are discussed. Due to rarity of this pathological condition, there are no guidelines concerning treatment and follow-up. We present our follow-up scheme and highlight the use of laparoscopic partial cystectomy as a successful treatment approach.

Download Full-text

CONGENITAL PTERIONAL DERMAL SINUS IN AN 18-MONTH-OLD CHILD

Neurosurgery ◽

10.1227/01.neu.0000290921.93588.87 ◽

2007 ◽

Vol 61 (3) ◽

pp. E661-E661 ◽

Cited By ~ 5

Author(s):

William J. Mack ◽

Saadi Ghatan

Keyword(s):

Clinical Features ◽

Neural Tube ◽

Clinical Presentation ◽

Sinus Tract ◽

Dermal Sinus ◽

Operating Microscope ◽

First Case ◽

Dermal Sinus Tract ◽

Cranial Fossa ◽

Congenital Dermal Sinus

Abstract OBJECTIVE Congenital dermal sinus tracts most frequently occur in the midline and are often associated with dermoid or epidermoid inclusion cysts. A lateral cranial origin is extremely rare and anatomically inconsistent with an etiology involving closure of the rostral neural tube during embryogenesis. CLINICAL PRESENTATION We describe the first case of a pterional cranial dermal sinus tract associated with a small epidermoid, with extension to the dura of the anterior cranial fossa. INTERVENTION The extra- and intracranial portions of the tract were visualized and resected with the use of an operating microscope. Pathological diagnosis confirmed dermal sinus tract. CONCLUSION We review the existing literature and address the developmental and clinical features pertinent to the management of cranial dermal sinus tracts, emphasizing the need for exploration of these potentially harmful lesions.

Download Full-text

Melioidosis in Bihar, India: unearthing the first of many?

Access Microbiology ◽

10.1099/acmi.0.000260 ◽

2021 ◽

Vol 3 (9) ◽

Author(s):

Isra Halim ◽

Prathyusha Kokkayil ◽

Ravi Kirti ◽

Rajeev Nayan Priyadarshi ◽

Asim Sarfraz ◽

...

Keyword(s):

Clinical Manifestations ◽

Clinical Suspicion ◽

Radiological Features ◽

First Case ◽

Insidious Onset ◽

Ankle Joints ◽

The World ◽

Resource Poor ◽

Empirical Antibiotics ◽

The Right

Melioidosis, a disease with protean clinical manifestations, is prevalent in many parts of India, with established endemic hotspots on the southern coast of the country. However, it is still underdiagnosed in many resource-poor regions of the country. We report what is, to the best of our knowledge, the first case of melioidosis diagnosed and treated in Bihar, an economically underdeveloped state in East India. The patient, a 52-year-old diabetic male, presented to the outpatient department with a fever of insidious onset along with pain and restriction of movement in the right shoulder joint and right knee joint, and swelling and tenderness of bilateral ankle joints. Radiological features were suggestive of multiple joint and organ abscesses. A diagnosis of disseminated septicaemic melioidosis was confirmed microbiologically. The patient improved clinically following aggressive treatment with meropenem and cotrimoxazole. The case highlights the need for increased clinical suspicion of melioidosis and adequate diagnostic facilities, as well as the need for early institution of appropriate empirical antibiotics in suspected cases of melioidosis in this region of the world.

Download Full-text

Spontaneous cervical epidural hematoma: Insight into this occurrence with case examples

Surgical Neurology International ◽

10.25259/sni_15_2021 ◽

2021 ◽

Vol 12 ◽

pp. 79

Author(s):

Brian Fiani ◽

Ryan Jarrah ◽

Nicholas J. Fiani ◽

Juliana Runnels

Keyword(s):

Epidural Hematoma ◽

Clinical Presentation ◽

Treatment Algorithm ◽

Region Of Interest ◽

Sudden Onset ◽

Neurological Deficits ◽

Spinal Epidural Hematoma ◽

Comprehensive Overview ◽

Case Examples ◽

First Case

Background: First characterized in the 19th century, spontaneous spinal epidural hematoma (SSEH) is known as the idiopathic accumulation of blood within the spinal canal’s epidural space, causing symptoms varying from general back pain to complete paraplegia. With varying etiologies, a broad spectrum of severity and symptoms, a time-dependent resolution period, and no documented diagnosis or treatment algorithm, SSEH is a commonly misunderstood condition associated with increasing morbidity. While SSEH can occur at any vertebrae level, 16% of all SSEH cases occur in the cervical spine, making it a region of interest to clinicians. Case Description: Herein, the authors present two case examples describing the clinical presentation of SSEH, while also reviewing the literature to provide a comprehensive overview of its presentation, pathology, and treatment. The first case is a patient with nontraumatic sudden onset neck pain with rapidly progressing weakness. The second case is a patient with painless weakness that developed while taking 325 mg of aspirin daily. Conclusion: Clinicians should keep SSEH in their differential diagnosis when seeing patients with nontraumatic sources of weakness in their extremities. The appropriate steps should be followed to diagnose and treat this condition with magnetic resonance imaging and surgical decompression if there are progressive neurological deficits. There is a continued need for more extensive database-driven studies to understand better SSEHs clinical presentation, etiology, and ultimate treatment.

Download Full-text

Refractory Thrombocytopenia Associated to Disseminated Histoplasmosis in a Living-Related Renal Transplant Recipient: A Case Report and Review of the Literature

Austin Journal of Clinical Case Reports ◽

10.26420/austinjclincaserep.2021.1218 ◽

2021 ◽

Vol 8 (6) ◽

Author(s):

Rosario MD ◽

◽

Alfredo G-G ◽

De Jesus AOA ◽

Jorge AS ◽

...

Keyword(s):

Renal Transplant ◽

Transplant Recipient ◽

Renal Transplant Recipient ◽

Clinical Characteristics ◽

Clinical Presentation ◽

Opportunistic Infections ◽

Review Of The Literature ◽

Disseminated Histoplasmosis ◽

First Case ◽

Living Related

Opportunistic infections are frequent complications after renal transplantation because of the use of immunosuppressants. Disseminated Histoplasmosis (DH) is one such opportunistic infection, and its clinical presentation varies, which makes its diagnosis a challenge. There is no information regarding DH as a cause of refractory thrombocytopenia in renal transplant recipient (RTR); therefore, we consider this an atypical case, and, because of its clinical characteristics, we have classified it as an Immune Thrombocytopenic Purpura (ITP) induced by histoplasmosis. This is the first case reported in our milieu, and it opens up the possibility for use of intravenous immunoglobulin as a strategic therapy for thrombocytopenia induced by HP in immunosuppressed RTRs.

Download Full-text

A Case of Liver Abscess withDesulfovibrio desulfuricansBacteremia

Case Reports in Infectious Diseases ◽

10.1155/2015/354168 ◽

2015 ◽

Vol 2015 ◽

pp. 1-4 ◽

Cited By ~ 3

Author(s):

Saho Koyano ◽

Keita Tatsuno ◽

Mitsuhiro Okazaki ◽

Kiyofumi Ohkusu ◽

Takashi Sasaki ◽

...

Keyword(s):

Liver Abscess ◽

Clinical Presentation ◽

Anaerobic Bacteria ◽

Desulfovibrio Desulfuricans ◽

Clinical Specimens ◽

Sulfate Reducing ◽

Oral Amoxicillin ◽

First Case ◽

Causative Agents ◽

Amoxicillin Clavulanic Acid

Desulfovibriospp. are gram-negative, sulfate-reducing, and anaerobic bacteria found in the digestive tract of humans. BecauseDesulfovibriospp. are infrequent causative agents of infectious diseases and are difficult to isolate and to identify from clinical specimens, the appropriate antibiotic therapy to infection withDesulfovibriospp. has not been determined. We report the first case of liver abscess with bacteremia due toDesulfovibrio desulfuricansto show the clinical presentation and treatment. The patient was successfully treated with intravenous piperacillin-tazobactam and oral amoxicillin-clavulanic acid.

Download Full-text