bluuebunny/crossref_metadata_2025 huggingface dataset:
Primary squamous cell carcinoma of the testis: a rare presentation
Authors: Zahoor Aqsa , Swamy Rajiv , Gogbashian Andrew , Sharma Anand
Published: May 2022

A man in his mid-70s presented with a lump in his left testicle. He had previously been treated for prostate cancer with radical radiotherapy. He was on treatment for hypertension and type 2 diabetes. An ultrasound of the testes demonstrated a solid intratesticular mass for which he underwent left orchidectomy. Histology from the orchidectomy was moderately differentiated squamous cell carcinoma (SCC), positive for cytokeratin (CK) 5/6 and p63. A positron emission tomogram (PET) scan was clear of any metastatic disease. His surveillance CT, done at 12 months, revealed mediastinal, abdominal and hilar adenopathy. Biopsy of hilar lymph nodes showed SCC and this was treated with platinum-based chemotherapy. Unfortunately, the patient died after 18 months. To our knowledge, this is the first reported case of metastatic SCC of testes with extensive spread and with platinum-refractory disease.
Case of GPD1 deficiency causing hypertriglyceridaemia and non-alcoholic steatohepatitis
Authors: Polchar Leo , Vallabhaneni Pramodh
Published: May 2022

We report a case of a young girl of South Asian descent presented with faltering growth, hepatomegaly, hypertriglyceridaemia and raised transaminases. Subsequent ultrasound scans identified fatty infiltration in her liver, and a liver biopsy showed fibrosis and steatosis. The patient’s serum triglycerides normalised without intervention by the age of 28 months. At age 6, whole-exome sequencing of the patient’s genome identified novel homozygous variants in the glycerol-3-phosphate dehydrogenase 1 (GPD1) gene,GPD1c.500G>A (p.Gly167Asp), leading to a diagnosis of GPD1 deficiency.
A rare and complete response to combination therapy with radiation and nivolumab in a patient with metastatic urothelial cancer
Authors: Hietala Aki , Joutsen Jenny , Vaarala Svea , Säily Matti
Published: May 2022

According to the current understanding, radiotherapy can enhance the effectiveness of cancer immunotherapy due to radiation-induced release of tumour-associated antigens. Here, we present a case with a metastatic urothelial carcinoma who received nivolumab and palliative radiotherapy to a residual tumour in the vagina and to a large metastatic visceral lymph node. The treatment resulted in a rapid and virtually complete response for the time being in all metastases and in the large parailiac tumour mass. Follow up continues. The presented case demonstrates that the combinatory treatment with radiotherapy and immunotherapy can result in an exceptional response for the benefit of the patient with urothelial cancer. To our knowledge, this is one of the largest metastatic masses to disappear with a combination of immuno-oncologic (nivolumab) and radiation therapies.
Early renal failure in childhood in a male with Fabry disease
Authors: Hogh Josephine Norre , Ebrahim Hatim , Moochhala Shabbir , Ramaswami Uma
Published: May 2022

Fabry disease is an X-linked lysosomal storage disorder caused by reduced activity or absence of the alpha-galactosidase A enzyme resulting in systemic accumulation of glycosphingolipids. End-stage renal disease (ESRD) is a late-stage manifestation of Fabry disease, typically presenting in the fifth decade of life, but is very rare in childhood. Here we present a case of an 11-year-old boy with classical Fabry disease presenting with ESRD requiring haemodialysis and transplant. Diagnosis was confirmed by renal biopsy,GLAmutation and low alpha-galactosidase A levels. He has an unusual genotype, hemizygous for the c.1000–11T>A intronic variant and positive for the pseudodeficiency allele D313Y. Due to the possibility of very early and accelerated disease progression, Fabry disease should be considered as a possible diagnosis in unexplained renal failure in males from a younger age.
Osteosclerotic metaphyseal dysplasia, dysosteosclerosis or osteomyelitis? Paediatric case presentation with associated mandibular swelling and a review of the literature
Authors: Chundoo Shima , McGoldrick David M , Williams Rhodri , McMillan Kevin
Published: May 2022

Osteosclerotic metaphyseal dysplasia (OMD) is an extremely rare form of osteopetrosis, which bears significant clinical similarities to dysosteosclerosis (DSS). We aim to present a rare case of OMD with mandibular swelling and osteomyelitis infection including diagnosis journey as well as management in 7-year-old patient. Literature review completed for OMD cases. Case report investigative methods include genetic testing, CT facial bones and MRI scan, orthopantogram and bone biopsies. An initial suspected diagnosis of DSS with chronic osteomyelitis was made. However, following genetic testing, a diagnosis of OMD was confirmed. Our patient underwent a surgical debulking procedure and antibiotic treatment. Less than 10 patients with this condition have been reported within the international literature. There is a wide range of presentation. OMD, DSS and osteomyelitis are all within a similar spectrum of bone conditions. Our understanding, regarding OMD, remains limited and, hence, further research is required to elucidate a thorough clinical picture.
Erythema multiforme in a young adult following COVID-19 infection and vaccination in Tanzania
Authors: Manji Karim Premji , Mwaniki George , Simba Alma Abu , Makani Julie
Published: May 2022

We report the case of a young female adult in her early 20s, who had COVID-19 infection for 8 weeks and COVID-19 vaccination 4 weeks prior to presentation with an extensive rash associated with erythema multiforme, resembling varicella zoster on initial presentation. After initial acyclovir therapy with no improvement, systemic corticosteroid treatment dramatically resolved the patient’s skin rash.
Perianal abscess as a manifestation of vaginocutaneous fistula after pelvic floor reconstruction
Authors: Safadi Mhd Firas , Berger Matthias
Published: May 2022

We present a case of vaginocutaneous fistula 10 years after pelvic floor reconstruction using transvaginal mesh implantation. The female patient in her mid-60s presented with typical symptoms of perianal abscess. After undergoing three surgical operations, the perianal infection was shown to be due to the implanted mesh as a late complication of the reconstruction. The patient was successfully managed with limited removal of the mesh tails. The case highlights one of the late complications of pelvic floor reconstruction using synthetic meshes.
Papillitis and uveitis complicating Bacillus Calmette-Guérin immunotherapy
Authors: Toohey Thomas P , Wallace Sharon , Toohey Michael G , Francis Ian C
Published: May 2022

Intravesical Bacillus Calmette-Guérin (BCG) administration was used to treat bladder carcinoma in a woman in her 60s. Severe bilateral non-granulomatous anterior uveitis and gross papillitis developed subsequently. The severe BCG-induced bilateral uveitis and papillitis were treated with high dose oral corticosteroids, with topical steroids and cycloplegics. Resolution of her ocular symptoms and signs eventuated. On lumbar puncture, no evidence of systemic spread of the BCG was found. Visual acuity returned to 6/9 in each eye with subsequent resolution of papillitis. Repeat cystoscopy demonstrated no evidence of recurrent bladder tumour.

Hypersensitivity reactions are well recognised withTubercle bacilli. While both hypersensitivity reactions and dissemination of BCG throughout the body have been previously documented, the literature demonstrates that this case is the first example in which papillitis and bilateral uveitis were the prominent ophthalmological features.
Metastatic follicular carcinoma arising from struma ovarii
Authors: Deng Jia , Ng Yang Huang Grace , Chew Sung Hock , Lim Yong Kuei
Published: May 2022

Struma ovarii (SO) is an uncommon monodermal teratoma predominantly composed of mature thyroid tissue. Approximately 5% of SO are malignant; however, metastases are rare. A single female in her 40s, with a medical history of Graves’ disease and bilateral cystectomy 10 years prior for right endometriotic cyst and left SO, presented with an enlarging abdominal mass for 4 months. Ultrasound pelvis showed a 13.8 cm left adnexal heterogeneous solid-cystic mass with internal septations and vascularity. She underwent open left salpingo-oophorectomy and resection of fibrous nodules from the right infundibulo-pelvic ligament and fallopian tube. Histology showed highly differentiated metastatic follicular carcinoma. She subsequently underwent total thyroidectomy, total hysterectomy, right salpingo-oophorectomy, tumour debulking and omentectomy followed by radioactive iodine treatment. Four-year follow-up did not show tumour recurrence or metastases. Due to its rarity, there are no well-established guidelines for the management and follow-up of metastatic follicular carcinoma arising from SO.
Tongue graft-versus-host disease: remission with ruxolitinib
Authors: Grafanaki Katerina , Lygeros Spyridon , Spyridonidis Alexandros , Liga Maria
Published: May 2022

Graft-versus-host disease (GvHD) is a potentially life-threatening and commonly encountered event of allogeneic haematopoietic stem cell transplantation. Here, we present a young adult male with primary refractory Hodgkin’s lymphoma who received a transplant and developed cutaneous GvHD after donor lymphocyte infusion, which was managed with cyclosporine and steroids. However, while the patient was under immunosuppressive treatment, diffuse confluent whitish patches on the patient’s tongue were observed. A biopsy of the tongue lesions revealed lichenoid, hyperkeratotic tissue changes and intraepithelial T-cell infiltration consistent with chronic GvHD. He was treated with mycophenolate mofetil for 6 months with minimal improvement. Janus-associated kinase inhibitor (ruxolitinib) treatment was commenced, with complete resolution of the tongue lesions and treatment discontinuation 5 months later. Currently, 5 years after allogeneic transplantation, he is in remission and does not need immunosuppressive therapy.
Multi-staged management of a mangled, exploded hand crush injury with long-term outcome: the critical link between surgery and therapy
Authors: Anderson Spencer R , Vincent Jacob A , Wimalawansa Sunishka M
Published: May 2022

A 38-year-old right-hand dominant man sustained a severe, work-related left-hand crushing injury. A multi-staged approach to salvage was employed in conjunction with aggressive hand therapy involvement for a successful, functional outcome. Now at approximately 5 years postinjury, the patient can perform daily activities, coaches sports and has returned to and maintained his full-time work position. Mangled, severe hand crush injuries warrant immediate treatment to debride non-salvageable tissues, stabilise and revascularise the hand in an effort to maximise reconstructive potential and functional capacity. It is critical to recognise that these cases require multiple stages of operative reconstruction with direct and ongoing involvement of hand therapy and pending degree of injury, rehabilitation often lasting months to years.
Oncocytic adrenocortical neoplasm with undetermined malignant potential and autonomous cortisol secretion
Authors: San Juan Mari Des , Lagamayo Dian , Carnate Jose , Joven Mark Henry
Published: May 2022

Increasing incidence of adrenal incidentalomas with hypercortisolemia not associated with overt features of Cushing’s syndrome has led to the evolution of the term autonomous cortisol secretion. Oncocytic adrenocortical neoplasms (OANs) are rare adrenal cortex neoplasms with only 250 reported cases worldwide. We present a woman in her 30s with menometrorrhagia, weight gain and increasing abdominal girth who was found to have a large right adrenal mass on abdominal CT scan. Serum cortisol was not suppressed after 1 mg dexamethasone (31.5 µg/dL) and adrenocorticotrophic hormone was not detectable (<1.0 pg/mL). She underwent right adrenalectomy under glucocorticoid coverage. Hypertension, weight, visceral adiposity and menometrorrhagia improved postoperatively. Histopathology with immunohistochemistry showed OAN with undetermined malignant potential. These kinds of tumours can only be definitively diagnosed postsurgically using the Lin-Weiss-Bisceglia system. Although they have better prognosis compared with adrenocortical carcinomas and do not require chemotherapy, patients should be closely monitored to identify recurrence promptly.
Filariasis presenting as pancytopenia in a child
Authors: Mandal Anusree Krishna , Jana Jadab Kumar , Gayen Soumya , Pal Abhay Charan
Published: May 2022

A female child hailing from South Asia, India presented with pallor, multiple petechiae and ecchymosis. Based on the clinical picture and demography, the differentials considered were pancytopenia of nutritional origin, acute leukaemia, autoimmune and infective aetiologies. After ruling these out by respective tests, a literature review was done which revealed the possibility of filariasis especially in a patient with eosinophilia which was present in our case. A repeat peripheral blood smear study with a nocturnally drawn sample revealed multiple microfilariae and a diagnosis of filariasis was made. The patient was treated with triple drug therapy of diethylcarbamazine (6 mg/kg), ivermectin (6 µg/kg) and albendazole (400 mg) administered as a single dose. Subsequent haemograms showed improved cell counts. This along with a previous handful of case reports emphasises filariasis as one of the differentials of pancytopenia and should be kept in mind while evaluating for the same, especially in the endemic areas.
Acute herpes zoster radiculopathy mimicking cervical radiculopathy after ChAdOx1 nCoV-19/AZD1222 vaccination
Authors: Dermawan Andrew , Ting Matthew Jun Min , Chemmanam Thomas , Lui Chok
Published: May 2022

This case report describes an 84-year-old man who presented with 3 weeks of gradually worsening right arm weakness associated with a painful vesicular rash across his arm. This occurred 3 days after his first dose of the ChAdOx1 nCoV-19/AZD1222 (University of Oxford, AstraZeneca and the Serum Institute of India) vaccine. The diagnosis was complicated by the presence of right C5–C6 foraminal stenosis compressing on the C6 nerve root sheath on non-contrast MRI, leading to an initial diagnosis of cervical radiculopathy. However, a positive varicella zoster virus-PCR test and findings of abnormal contrast enhancement of his right C5–C7 nerve roots on gadolinium-enhanced MRI resulted in a revision of his diagnosis to zoster radiculopathy. He was subsequently commenced on oral valacilovir and made significant recovery. This report aims to highlight the diagnostic dilemma between cervical radiculopathy secondary to spondylosis and zoster radiculopathy and how an erroneous diagnosis could result in inappropriate, aggressive surgical intervention and delayed treatment with antiviral therapy.
Maintenance therapy for long-term control of high-grade endometrial stromal sarcoma
Authors: Nakanishi Kazuho , Maeoka Yuri , Yamada Takashi , Suzuki Shunji
Published: May 2022

High-grade endometrial stromal sarcoma (HG-ESS) is a malignant uterine tumour with a poor prognosis. This study aimed to demonstrate the usefulness of maintenance chemotherapy with paclitaxel (PTX) and carboplatin (CBDCA) for HG-ESS recurrence, by examining a case of HG-ESS tumour progression as a result of maintenance therapy interruption because of the COVID-19 pandemic. The patient was a woman in her 60s who presented with lower abdominal pain, nausea and a pelvic tumour, which was diagnosed as HG-ESS. Chemotherapy with PTX (175 mg/m2day 1) + CBDCA (area under the concentration-time curve 5 day 1) every 21–28 days (TC therapy) was administered for long-term maintenance of HG-ESS. After 26 cycles, chemotherapy had to be postponed because of the COVID-19 pandemic, thus leading to disease progression and eventually death. In conclusion, TC therapy can prolong survival in patients with HG-ESS, and maintenance chemotherapy should not be postponed.
Rhombencephalomyelitis due to possible paraneoplastic syndrome associated with Hodgkin’s lymphoma
Authors: Valappil Ashraf , Radhakrishnan Nair Ragesh , Madhusoodhanan Sudha , Narendran Anoop
Published: May 2022

We report a unique case of rhombencephalomyelitis with unclear aetiology, diagnosed with Hodgkin’s lymphoma (HL) on follow-up.

A woman in her 50s was presented with gait difficulty, dysarthria, left Horner’s syndrome and left trigeminal sensory loss. MRI of the brain revealed an enhanced T2 and fluid attenuated inversion recovery hyperintense lesion at the cervico-medullary junction, suggesting rhombencephalomyelitis. The initial differential diagnosis included demyelination, other inflammatory conditions like sarcoidosis, Behcet’s disease and paraneoplastic syndrome. Cerebrospinal fluid analysis, serum myelin oligodendrocyte glycoprotein, neuromyelitis optica antibodies, vasculitis work-up, onconeural antibody panel and whole-body fluorodeoxyglucose-positron emission tomography scan were all unrevealing, and she was kept under close follow-up. After 18 months, she developed a prolonged fever and was diagnosed with HL. Rhombencephalomyelitis as a HL-associated paraneoplastic syndrome has not been reported in the literature. This case report highlights the importance of serial screening for occult malignancy in patients with suspected paraneoplastic syndrome.
Roifman syndrome: a description of further immunological and radiological features
Authors: Clifford Danielle , Moloney Fiachra , Leahy Timothy Ronan , Murray Deirdre M
Published: May 2022

Roifman syndrome is a rare autosomal recessive inherited syndromic immunodeficiency. We wish to add to the available literature by reporting two brothers with clinical, radiological and immunological features of Roifman syndrome, confirmed on whole exome sequencing. We report an excellent response to subcutaneous immunoglobulin therapy in both brothers, reducing infection burden and hospital admissions. New radiological features are also described here which may assist in the diagnosis of other patients.
Rare case of mesocardia with ostium secundum ASD and double IVC
Authors: Kumar Shriram , Nandyal Shreyas Raghavan , Kaushik Sharanya , Ibrahim Sufyan
Published: May 2022

We report an extremely rare case of mesocardia with double inferior vena cava (IVC) in this case report. The patient’s associated atrial septal defect (ASD) with left to right shunt leading to pulmonary congestion and recurrent episodes of pneumonia was the leading diagnosis for which care was sought at our hospital. The diagnoses of these rare congenital cardiac anomalies were uncovered only after a thorough workup. Mesocardia is extremely rare and accounts for only 0.2% of congenital anomalies. Its association with other cardiac and vascular anomalies, however, warrants a meticulous workup for the identification of such associated conditions—ASD and double IVC being two such anomalies in our patient. The dictum of working up a case of congenital cardiac anomaly for other congenital cardiaovascular anomalies, therefore, found merit in our case.
Giant renal parapelvic cyst with pelvi-ureteric junction obstruction in an infant: challenges in diagnosis and laparoscopic management
Authors: Kapoor Rohit , Mandelia Ankur , Verma Anju , Kanneganti Pujana
Published: May 2022

Renal parapelvic cysts (RPC) have an incidence of approximately 1%–3% in the general population. However, they rarely present in children with only two cases reported in literature. RPC are often misdiagnosed as it is difficult to distinguish them from hydronephrosis on preoperative imaging. We report a case of an infant with a giant RPC with associated pelvi-ureteric junction obstruction, who was managed successfully with laparoscopic complete excision of RPC and dismembered pyeloplasty. In our report, we discuss the challenges faced in the diagnosis and surgical management of this unusual case.
Multimodal imaging of congenital retinal macrovessel with secondary foveal hypoplasia
Authors: Kohli Piyush , Mishra Chitaranjan , Baliga Girish , Rajan Renu P
Published: May 2022

Congenital retinal macrovessel (CRM) is defined as a large aberrant blood vessel that traverses through the central macula. It can have large tributaries extending on both sides of the horizontal raphe. The condition is typically asymptomatic. However, visual loss may be reported in case of associated branch retinal artery occlusion, cavernous haemangioma, retinal artery macroaneurysm, retinal deep capillary ischaemia, retinal detachment and vitreous haemorrhage. However, association of CRM and foveal hypoplasia has rarely been reported.

We report multimodal imaging in a patient who was presented with visual loss due to foveal hypoplasia secondary to CRM.
Rare occurrence of Guillain-Barré syndrome after Moderna vaccine
Authors: Bijoy George Tissa , Kainat Aleesha , Pachika Pranali Santhoshini , Arnold Jonathan
Published: May 2022

A Caucasian man in his 60s with a medical history significant for ruptured left middle cerebral artery aneurysm status post clipping 2005 with residual right eye blindness and right leg weakness with gait instability presented with loss of balance, weakness of his legs and fatigue for 3 days. No other antecedent event was identified other than receiving Moderna COVID-19 vaccine 4 weeks before the presentation and 3 days before symptom onset. CT head and CT angiogram of the head and neck were performed and demonstrated no acute intracranial bleeding and no vascular abnormalities. With the findings of diffuse hyporeflexia and cerebrospinal fluid showing albumino-cytological dissociation, Guillain-Barré syndrome was high on the differentials. Electromyogram showed evidence of demyelination. He was treated with intravenous immune globulin (IVIG) and was discharged to rehab with complete symptom resolution.
Ovarian steroid cell tumour inducing virilisation in a postmenopausal woman
Authors: Pinto Ana Marta , Martins Maria Boia , Oliveira Nuno , Oliveira Mário
Published: May 2022

Hyperandrogenism with virilisation de novo in postmenopausal women is exceedingly rare, with aetiology oscillating between ovarian tumours, adrenal tumours, ovarian hyperthecosis and, less frequently, Cushing’s syndrome. We report a case of a postmenopausal woman in her late 60s, referred from her primary healthcare physician to a gynaecology appointment due to hirsutism and vasomotor symptoms. At physical examination, clitoromegaly was also identified. Blood tests revealed severe hyperandrogenemia, with total testosterone above 200 ng/dL, but transvaginal ultrasound and abdominal CT were unremarkable. Three months later, abdominal CT was repeated, revealing a moderate heterogeneous enhancement with 18 mm on the left ovary, which was confirmed by transvaginal ultrasound. Total laparoscopic hysterectomy with bilateral adnexectomy was performed. Histopathological examination reported an ovarian steroid cell tumour not otherwise specified on the left ovary and bilateral ovarian hyperthecosis. Two months later, the patient had normal total testosterone and the hirsutism complaints were completely absent.
Factors associated with glaucoma-specific quality of life in a US glaucoma clinic in a pilot implementation of an online computerised adaptive test (GlauCAT)
Authors: Halawa Omar A , Roldan Ana M , Meshkin Ryan S , Zebardast Nazlee , Fenwick Eva K , Lamoureux Ecosse Luc , Friedman David S
Published: May 2022

Objectives

Measure quality of life (QoL) outcomes using a novel computerised adaptive test in a clinical setting, and determine the social and demographic factors associated with specific QoL domains in patients with glaucoma.

Design

Cross-sectional study between July 2020 and April 2021.

Participants

English-speaking adults presenting to glaucoma clinic. Patients with cognitive impairment on a six-item cognitive impairment screen or with intraocular surgery within 90 days prior to presentation were excluded.

Results

Of 206 patients surveyed, mean age was 64.8 years (SD 15.2), 122 (56.7%) were female and 159 (74.7%) were white. On multivariable regression, visual acuity was associated with greater activity limitation (β=−2.8 points, 95% CI −3.8 to –1.8, p<0.001) and worse mobility (β=−2.1 points, 95% CI −3.2 to –0.9, p<0.001), while poorer visual field (VF) mean deviation was associated with lower scores on the emotional well-being domain (β=−2.4 points, 95% CI −4.6 to –0.3, p=0.03). Glaucoma suspects and those with early VF defects had higher QoL scores than those with severe glaucoma in the following domains: activity limitation (88.5±14.6 vs 74.3±21.9, respectively, p<0.001), mobility (91.0±12.5 vs 80.0±25.3, respectively, p=0.005) and concerns domains (82.2±13.9 vs 72.5 5±18.9, respectively, p=0.01).

Conclusions

In a busy glaucoma clinic where QoL was measured with online adaptive tests for glaucoma, we found that several demographic and clinical variables are associated with lower domain scores, suggesting that patients with predisposing demographic and clinical factors are at a higher risk of worse QoL.
Effects of exercise training on the cognitive function of older adults with different types of dementia: a systematic review and meta-analysis
Authors: Balbim Guilherme Moraes , Falck Ryan S , Barha Cindy K , Starkey Samantha Y , Bullock Alexis , Davis Jennifer C , Liu-Ambrose Teresa
Published: May 2022

Objectives

To assess the effect of exercise training on the cognitive function of older adults living with different types of dementia, as well as potential moderators of exercise efficacy.

Design

Systematic review and meta-analysis.

Data sources

Cochrane Central, PsycINFO, Embase, Medline and CINAHL.

Eligibility criteria

Peer-reviewed, randomised controlled trials, in English (1990–present), which examined the effects of exercise training on the cognitive function of older adults living with dementia.

Study appraisal and synthesis

Risk of bias and study quality were assessed (Cochrane Risk of Bias Tool 2.0 and Physiotherapy Evidence Database Scale). We performed random-effects models using robust variance estimation and tested moderators using the approximate Hotelling-Zhang test.

Results

Twenty-eight studies (n=2158) were included in the qualitative review and 25 in the meta-analysis. For all-cause dementia, a small effect of exercise training on cognitive function was observed (g=0.19; 95% CI 0.05 to 0.33; p=0.009). Type of dementia and exercise training characteristics did not moderate the effects of exercise training on cognitive function (p>0.05). Adherence to the intervention moderated the cognitive outcome effect size such that greater mean adherence was associated with greater cognitive outcome effect sizes (b=0.02;SE=0.01; p=0.005).

Conclusion

Exercise training showed small benefits for the cognitive function of older adults living with all-cause dementia. More research and standardised reporting of exercise training characteristics can strengthen the evidence for what works best for which types of dementia.

PROSPERO registration number

CRD42020198716.
When do people with abdominal symptoms need a fast track referral?
Authors: Saul Helen , Gursul Deniz , Herbert Annie , Lyratzopoulos Georgios
Published: May 2022

The study

Herbert A, Rafiq M, Pham TM, et al. Predictive values for different cancers and inflammatory bowel disease of 6 common abdominal symptoms among more than 1.9 million primary care patients in the UK: a cohort study.PLoS Med2021:18;e1003708.

To read the full NIHR Alert, go to:https://evidence.nihr.ac.uk/alert/combining-risks-cancer-and-ibd-allows-more-fast-track-referrals/
Women with type 2 diabetes and coronary artery disease have a higher risk of heart failure than men, with a significant gender interaction between heart failure risk and risk factor management: a retrospective registry study
Authors: Fujita Yuko , Morimoto Takeshi , Tokushige Akihiro , Ikeda Masayuki , Shimabukuro Michio , Node Koichi , Ueda Shinichiro
Published: May 2022

Introduction

Women are generally perceived to have a lower risk of cardiovascular events than men, despite a lack of data, particularly among patients with diabetes. Here, we investigated gender differences in the risk of heart failure (HF) events in patients with type 2 diabetes and coronary artery disease (CAD). We also assessed the association between cardiovascular risk factor management and HF events.

Research design and methods

This retrospective registry study enrolled consecutive patients with both type 2 diabetes and CAD, based on angiography records and medical charts at 70 teaching hospitals in Japan, from January 2005 to December 2015.

Results

The registry included 7785 patients with a mean follow-up period of 1328 days. The mean age of the patients was 67.6 years. The risk of hospitalization for HF in patients with both type 2 diabetes and CAD was significantly higher among women than among men (HR, 1.26, 95% CI 1.06 to 1.50). The relationship between HF risk and achieved low-density lipoprotein cholesterol (LDL-c) and systolic blood pressure, but not hemoglobin A1c, differed between women and men, with statistically significant interactions (p=0.009 and p=0.043, respectively).

Conclusions

Women with type 2 diabetes and CAD have a higher risk of HF than men. A significant gender interaction was observed in the association between HF risk and risk factor management, particularly regarding LDL-c and systolic blood pressure. The effectiveness of risk factor management may differ between men and women regarding HF prevention among patients with type 2 diabetes and CAD.
Reporting of patient-reported outcomes in trials on alcohol use disorder: a meta-epidemiological study
Authors: Douglas Alexander , Garrett Elizabeth , Staggs Jordan , Williams Cole , Shepard Samuel , Wise Audrey , Hillman Cody , Ottwell Ryan , Hartwell Micah , Vassar Matt
Published: May 2022

Objectives

Currently, limited research exists to assess the extent of patient-reported outcome (PRO) reporting among randomised controlled trials (RCTs) evaluating alcohol use disorder (AUD). We sought to investigate the completeness of reporting of PROs using the Consolidated Standards of Reporting Trials-PRO (CONSORT-PRO) extension in AUD RCTs.

Design setting

Meta-epidemiological study.

Methods

We searched MEDLINE, Embase and the Cochrane Central Register of Controlled Trials (CENTRAL) on 29 June 2021 for published RCTs focused on AUD. Following these searches, title and abstract screening, and full-text screening were performed by two investigators. To be included, a study must have employed a randomised trial design, published in English, focused on treatment of AUD and included at least one PRO. Trials meeting inclusion criteria were evaluated for completeness of reporting using the CONSORT-PRO extension adaptation. These trials were also evaluated for risk of bias (RoB) using the Cochrane RoB V.2.0 tool. Additionally, an exploratory analysis of each RCT’s therapeutic area was extracted using the Mapi Research Trust’s ePROVIDE platform. Screening and data collection were all performed in masked, duplicate fashion.

Main outcome measures

PRO completeness of reporting, identification of factors associated with completeness of reporting and PRO measures used in RCTs to evaluate patients with AUD.

Results

Nineteen RCTs were evaluated in our analysis. Our primary outcome, the mean completion score for CONSORT-PRO, was 40.8%. Our secondary outcome—the identification of factors associated with completeness of reporting—found that trials published after 2014 (ie, 1 year after the publication of the CONSORT-PRO extension) were 15.0% more complete than trials published before 2014. We found no additional associations with better reporting.

Conclusions

We found that the completeness of PRO reporting in RCTs involving AUD was deficient. Complete reporting of PROs is instrumental in understanding the effects of interventions, encourages patient participation in their treatment and may increase clinician confidence in the value of PROs. High quality treatment strategies for AUD require properly reported PROs.
FDA’s dilemma with the aducanumab approval: public pressure and hope, surrogate markers and efficacy, and possible next steps
Authors: Rizk John G , Lewin John C
Published: May 2022

Accelerating Food and Drug Administration (FDA) product approval to market based on surrogate markers in the absence of proven efficacy creates a risk of adverse outcomes for affected patients, even in response to a life-threatening condition, such as in this case, Alzheimer’s disease. FDA’s recent unexpected approval of aducanumab, despite the unified opposition of its own highly respected advisory committee after the early termination of two efficacy trials, creates the potential risk of adverse effects and lack of clinical efficacy at very high costs. In view of these concerns, a thorough review of the issues and pressures that led to this decision is worth the careful consideration of the clinical and scientific communities with regard to whether this approval represents a calculated and balanced compassionate decision versus a disturbing precedent.
Multimorbidity and associations with clinical outcomes in a middle-aged population in Iran: a longitudinal cohort study
Authors: Odland Maria Lisa , Ismail Samiha , Sepanlou Sadaf G , Poustchi Hossein , Sadjadi Alireza , Pourshams Akram , Marshall Tom , Witham Miles D , Malekzadeh Reza , Davies Justine I
Published: May 2022

Background

As the populations of lower-income and middle-income countries age, multimorbidity is increasing, but there is little information on its long-term consequences. We aimed to show associations between multimorbidity and outcomes of mortality and hospitalisation in Iran, a middle-income country undergoing rapid economic transition.

Methods

We conducted a secondary analysis of longitudinal data collected in the Golestan Cohort Study. Data on demographics, morbidities and lifestyle factors were collected at baseline, and information on hospitalisations or deaths was captured annually. Logistic regression was used to analyse the association between baseline multimorbidity and 10-year mortality, Cox-proportional hazard models to measure lifetime risk of mortality and zero-inflation models to investigate the association between hospitalisation and multimorbidity. Multimorbidity was classified as ≥2 conditions or number of conditions. Demographic, lifestyle and socioeconomic variables were included as covariables.

Results

The study recruited 50 045 participants aged 40–75 years between 2004 and 2008, 47 883 were available for analysis, 416 (57.3%) were female and 12 736 (27.94%) were multimorbid. The odds of dying at 10 years for multimorbidity defined as ≥2 conditions was 1.99 (95% CI 1.86 to 2.12, p<0.001), and it increased with increasing number of conditions (OR of 3.57; 95% CI 3.12 to 4.08, p<0.001 for ≥4 conditions). The survival analysis showed the hazard of death for those with ≥4 conditions was 3.06 (95% CI 2.74 to 3.43, p<0.001). The number of hospital admissions increased with number of conditions (OR of not being hospitalised of 0.36; 95% CI 0.31 to 0.52, p<0.001, for ≥4 conditions).

Conclusion

The long-terms effects of multimorbidity on mortality and hospitalisation are similar in this population to those seen in high-income countries.
Cash, COVID-19 and aid cuts: a mixed-method impact evaluation among South Sudanese refugees registered in Kiryandongo settlement, Uganda
Authors: Stein Daniel , Bergemann Rico , Lanthorn Heather , Kimani Emma , Nshakira-Rukundo Emmanuel , Li Yulei
Published: May 2022

Objectives

In this paper, we estimate the impact after 4–8 months of a large one-off unconditional cash transfer delivered to refugees during a time of dual shocks: the COVID-19 pandemic and cuts to monthly aid. We focus on four key outcomes: (1) health-seeking behaviour; (2) COVID-19 specific preventive health practices; (3) food security and (4) psychological well-being.

Methods

We use both quantitative and qualitative data to understand the impact of a cash transfer in this context. Quantitatively, we use a baseline survey of 1200 households (Q4 2019) and follow-up with three rounds of phone surveys in Q2 and Q3 2021, capturing at least half the sample in each round. We exploit an experimental variation in the timing of the cash transfer to assess the effect of the cash transfer through ordinary least squares regressions of intention to treat. Controlling for key baseline characteristics, we analyse the effect of the cash transfer on health access, COVID-19 health practices, food security and psychological well-being. Qualitatively, we make use of a longitudinal, small-n sample of refugee respondents, each of whom we interviewed up to 15 times between February and September 2020 to understand change over time and to go deeper into key topics.

Results

We do not find a statistically significant effect (6.2%, p=0.188) of receiving the cash transfer on preventative measures against COVID-19. However, households receiving the cash transfer were more food secure, with a 14.4% (p=0.011) improvement on the food security index, have better psychological well-being (24.5%, p=0.003) and are more likely to seek healthcare in the private health facilities (10.4%, p=0.057) as compared with control households. We do not find significant results on the value of food consumption. Overall, we find stronger treatment effects for households that were the first to receive the cash transfers.

Conclusion

Taken together, we find significant support for the importance of cash transfers to refugee households mitigating against declines in food security and mental well-being in the face of shocks.
Alarming level of severe acute malnutrition in Indian districts
Authors: Ulahannan Sabu Kochupurackal , Wilson Alby , Chhetri Deepshikha , Soman Biju , Prashanth NS
Published: May 2022

Over the last two decades, severe acute malnutrition (SAM) has been increasing in India despite favourable national-level economic growth. The latest round of the National Family Health Survey 5 (NFHS-5) results was released, allowing us to assess changes in the malnutrition trends. Analysis of the previous rounds of the NFHS (NFHS-4) has already shown disturbing levels of wasting, often co-occurring with other forms of anthropometric failures. These have been shown to occur in clusters of districts across India that already needed urgent policy and programmatic action. A rapid assessment of data from NFHS-5 for some of these districts for which data are now available shows an alarming increase in SAM in several malnutrition hotspot districts. Surprisingly, some districts outside hotspots and in states and regions that have previously not been known for high malnutrition too have shown increasing SAM prevalence in the latest round. The data from NFHS-5 was collected just before the COVID-19 pandemic and hence does not yet reflect the likely impact of the pandemic on food security, livelihoods and other social stressors among the most marginalised Indian households. Based on this emerging pattern of increasing SAM, we call for an urgent policy and programmatic action to strengthen the Anganwadi system, which caters to preschool children in India and community-based management of acute malnutrition based on recent evidence on their effectiveness.
Implications of COVID-19 for safeguarding in international development research: learning, action and reflection from a research hub
Authors: Mansaray Bintu , Dean Laura , Tubb Phil , Josyula Kunhi Lakshmi , Okoth Linet , Chumo Ivy , Waritu Jane , Klingel Andrea , Manzoor Farzana , Aktar Bachera , Garimella Surekha , Murthy Shrutika , Tolhurst Rachel , Whittaker Lana , Gray Linsay , Forsyth Ross , Elsey Helen , Waldman Linda , Theobald Sally
Published: May 2022

COVID-19 brings uncertainties and new precarities for communities and researchers, altering and amplifying relational vulnerabilities (vulnerabilities which emerge from relationships of unequal power and place those less powerful at risk of abuse and violence). Research approaches have changed too, with increasing use of remote data collection methods. These multiple changes necessitate new or adapted safeguarding responses. This practice piece shares practical learnings and resources on safeguarding from the Accountability for Informal Urban Equity hub, which uses participatory action research, aiming to catalyse change in approaches to enhancing accountability and improving the health and well-being of marginalised people living and working in informal urban spaces in Bangladesh, India, Kenya and Sierra Leone. We outline three new challenges that emerged in the context of the pandemic (1): exacerbated relational vulnerabilities and dilemmas for researchers in responding to increased reports of different forms of violence coupled with support services that were limited prior to the pandemic becoming barely functional or non-existent in some research sites, (2) the increased use of virtual and remote research methods, with implications for safeguarding and (3) new stress, anxiety and vulnerabilities experienced by researchers. We then outline our learning and recommended action points for addressing emerging challenges, linking practice to the mnemonic ‘the four Rs: recognise, respond, report, refer’. COVID-19 has intensified safeguarding risks. We stress the importance of communities, researchers and co-researchers engaging in dialogue and ongoing discussions of power and positionality, which are important to foster co-learning and co-production of safeguarding processes.
Building a national framework for multicentre research and clinical trials: experience from the Nigeria Implementation Science Alliance
Authors: Olawepo John Olajide , Ezeanolue Echezona Edozie , Ekenna Adanma , Ogunsola Olabanjo O , Itanyi Ijeoma Uchenna , Jedy-Agba Elima , Egbo Emmanuel , Onwuchekwa Chukwudi , Ezeonu Alexandra , Ajibola Abiola , Olakunde Babayemi O , Majekodunmi Omololuoye , Ogidi Amaka G , Chukwuorji JohnBosco , Lasebikan Nwamaka , Dakum Patrick , Okonkwo Prosper , Oyeledun Bolanle , Oko John , Khamofu Hadiza , Ikpeazu Akudo , Nwokwu Uchechukwu Emmanuel , Aliyu Gambo , Shittu Oladapo , Rositch Anne F , Powell Byron J , Conserve Donaldson F , Aarons Gregory A , Olutola Ayodotun
Published: May 2022

There is limited capacity and infrastructure in sub-Saharan Africa to conduct clinical trials for the identification of efficient and effective new prevention, diagnostic and treatment modalities to address the disproportionate burden of disease. This paper reports on the process to establish locally driven infrastructure for multicentre research and trials in Nigeria known as the Nigeria Implementation Science Alliance Model Innovation and Research Centres (NISA-MIRCs). We used a participatory approach to establish a research network of 21 high-volume health facilities selected from all 6 geopolitical zones in Nigeria capable of conducting clinical trials, implementation research using effectiveness-implementation hybrid designs and health system research. The NISA-MIRCs have a cumulative potential to recruit 60 000 women living with HIV and an age-matched cohort of HIV-uninfected women. We conducted a needs assessment, convened several stakeholder outreaches and engagement sessions, and established a governance structure. Additionally, we selected and trained a core research team, developed criteria for site selection, assessed site readiness for research and obtained ethical approval from a single national institutional review board. We used the Exploration, Preparation, Implementation, Sustainment framework to guide our reporting of the process in the development of this network. The NISA-MIRCs will provide a nationally representative infrastructure to initiate new studies, support collaborative research, inform policy decisions and thereby fill a significant research infrastructure gap in Africa’s most populous country.
The causal effect of delivery volume on severe maternal morbidity: an instrumental variable analysis in Sichuan, China
Authors: Chen Nan , Pan Jay
Published: May 2022

Objective

Findings regarding the association between delivery volume and maternal health outcomes are mixed, most of which explored their correlation. This study aims to demonstrate the causal effect of delivery volume on severe maternal morbidity (SMM) in China.

Methods

We analysed all women giving birth in the densely populated Sichuan province with 83 million residents in China, during the fourth quarters of each of 4 years (from 2016 to 2019). The routinely collected discharge data, the health institutional annual report data and road network data were used for analysis. The maternal health outcome was measured by SMM. Instrumental variable (IV) methods were applied for estimation, while the surrounding average number of delivery cases per institution was used as the instrument.

Results

The study included 4545 institution-years of data from 1456 distinct institutions with delivery services, reflecting 810 049 associated delivery cases. The average SMM rate was approximately 33.08 per 1000 deliveries during 2016 and 2019. More than 86% of delivery services were provided by a third of the institutions with the highest delivery volume (≥143 delivery cases quarterly). In contrast, less than 2% of delivery services were offered by a third of the institutions with the lowest delivery volume (<19 delivery cases quarterly). After adjusting the confounders in the IV-logistic models, the average marginal effect of per 1000 cases in delivery volume was −0.162 (95% CI −0.169 to –0.155), while the adjusted OR of delivery volume was 0.005 (95% CI 0.004 to 0.006).

Conclusion

Increased delivery volume has great potential to improve maternal health outcomes, while the centralisation of delivery services might facilitate maternal health promotion in China. Our study also provides implications for other developing countries confronted with similar challenges to China.
The Kenya UK Breast Cancer Awareness Week: curriculum codesign and codelivery with direct and lived experience of breast cancer diagnosis and management
Authors: Riogi Bahaty , Ross Carlo , Mutebi Miriam , Dave Rajiv V
Published: May 2022

Global health education holds a paradox: the provision of global health degrees focusing on challenges in low-income and middle-income countries has increased in high-income countries, while those in these low-income and middle-income countries lack access to contribute their expertise, creating an ‘information problem’. Breast cancer is a pressing global health priority, which requires curriculum design, implementation, ownership and leadership by those with direct and lived experience of breast cancer.

The Kenya-UK Breast Cancer Awareness Week was conceptualised following the signing of the Memorandum of Understanding between the Kenyan and UK governments launching the Kenya UK Health Alliance. This alliance aims to promote health cooperation to address Kenya’s breast cancer challenge. Here, we present the first of the collaborative’s initiatives: a breast cancer global health education programme designed, implemented, owned and led by Kenyan stakeholders.

We present the utilisation of the Virtual Roundtable for Collaborative Education Design for the design and implementation of a nationwide virtual breast cancer awareness week delivered across eleven Kenyan medical schools. By involving partners with lived and/or professional experience of breast cancer in Kenya in all stages of the design and delivery of the awareness week, the project experimented with disrupting power dynamics and fostered ownership of the initiative by colleagues with direct expertise of breast cancer in Kenya.

This initiative provides a platform, precedent and playbook to guide professionals from other specialties in the design and implementation of similar global collaborative ventures. We have used this approach to continue to advocate for global health curricula design change, so that those with lived experiences of global health challenges in their contextualised professional and personal environments are given leadership, reward and ownership of their curricula and further to highlight breast cancer as a global heath priority.