Sexual disorder improvement: a target or a way in treatment of fibromyalgia. A case report and brief review

Abstract

Previous studies had shown the relation between fibromyalgia (FM) and sexual impairment as a symptom of established disease, which causes often serious problems in partners’ relationship. We described a middle-aged man with FM who was refractory to conventional treatments after an 8-year history of generalized chronic pain. He underwent multiple treatment modalities, such as tricyclic antidepressants, selective serotonin reuptake inhibitors, serotonin?norepinephrine reuptake inhibitors, and nonpharmacological therapies, with no dramatic success. Psychiatric assessment revealed a homosexual tendency. He and his wife were informed about the problem. A change in sexual behavior caused a significant resolution of symptoms over a 6-month period, and he no longer receives medication for FM. This is the first case report to demonstrate the efficacy of sexual disorder improvement in the treatment of refractory FM.     

Sporadic amyotrophic lateral sclerosis with seropositive neuromyelitis optica spectrum disorder: A case report

Rationale:Neuromyelitis optica spectrum disorder (NMOSD) is a severe inflammatory disorder of the central nervous system with an autoantibody against aquaporin-4 protein (AQP4), and amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disease. We report a female patient with ALS who had asymptomatic AQP4 antibody at the diagnosis of ALS, and NMOSD occurred 4 years later after the diagnosis of ALS.Patient concerns:She was already bedridden and had tracheostomy because of ALS which was diagnosed at her age of 55. At the time of her ALS diagnosis, she had no brain or spinal cord lesions, but was seropositive for AQP4 antibody. At her age of 59, new-onset complete paralysis of all extremities and severe pain on the posterior neck and both shoulders occurred and visited the hospital.Diagnosis:Longitudinally extensive transverse myelitis was diagnosed, which was the onset attack of seropositive NMOSD. The diagnosis was confirmed based on the international consensus diagnostic criteria for NMOSD with MR imaging, cerebrospinal fluid exam and laboratory work-ups with AQP4 antibody test.Interventions:High dose methylprednisolone was administered for 5 days. Plasma exchange as a further treatment was recommended, but she and her family refused.Outcomes:Her pain was relieved after steroid treatment, but there was no improvement of her leg weakness.Lessons:This case is a rare combination of neuroinflammatory and neurodegenerative diseases. Considering the alterations of blood-brain barrier along with the progression of ALS, it highlights that the consequence of ALS pathogenesis might affect the development of NMOSD. And the careful follow-up is recommended even in patients with profound weakness, especially if those who were at risk of developing certain neurological disorders.     

N-methyl-d-aspartate receptor antibody encephalitis: a treatable disorder involving B-lymphocytes. A report of two patients

Introduction

N-methyl-D-aspartate receptor antibody (anti-NMDA-r AB) encephalitis has been recently identified. We report two cases illustrating the clinical features, response to immunomodulatory treatment and involvement of B-lymphocytes that characterizes this disorder.

Case reports

These patients illustrated the classic clinical features of anti-NMDA-r AB encephalitis including occurrence in young female, presence of severe neurological and psychiatric manifestations with confusion, seizures, mutism, hypovigilence and involuntary movements, and inflammatory cerebrospinal fluid. Both patients improved after immunotherapy. In case 1, the encephalitis was associated with an ovarian teratoma containing neuronal elements. In case 2, there was no tumor identified. A brain biopsy showed prominent perivascular B-cells infiltrates with some T-cells distributed in the brain parenchyma.

Conclusion

Anti-NMDA-r AB encephalitis is certainly not rare and needs to be promptly recognized and treated. An associated neoplasia is inconstant and the pathophysiology involves humoral immunity.     

Emotional responses to food in adults with an eating disorder: a qualitative exploration

There is a dearth of qualitative research exploring eating disordered individuals' emotional responses to the sight of different types of food. In this study we asked 10 adult women diagnosed with bulimia nervosa, anorexia nervosa or an eating disorder not otherwise specified (EDNOS) to articulate their thoughts whilst viewing slides of a range of different foodstuffs, during an in‐depth interview session. The data were transcribed and subjected to qualitative analysis. A core theme of ‘Control’ was identified. Whilst control has been previously acknowledged in the literature as an important aspect of eating disordered behaviour, this is the first report to link control with negative emotional responses to the sight of food. Clinically, an exploration of the notion of control and how it relates to particular foods may be beneficial in therapy. Copyright © 2007 John Wiley & Sons, Ltd and Eating Disorders Association.     

Transient myeloproliferative disorder in neonates without Down syndrome: case report and review

Transient myeloproliferative disorder (TMD) is a clonal proliferation of megakaryoblasts, typically occurring in newborns with Down syndrome. It is believed that TMD occurs in the presence of GATA1 mutation together with trisomy 21. However, a limited number of patients with TMD but without Down syndrome have been reported, all with a blast population with numeric or rarely structural chromosome 21 abnormalities. We present the first case of a newborn boy with a TMD without trisomy 21 and without any of the mentioned molecular or cytogenetic abnormalities. This case report suggests that unknown disease mechanisms may provoke or mimic TMD. This case report is followed by a concise review of the literature discussing the different entities and pathomechanisms of TMD and acute megakaryocytic leukaemia in patients with or without Down syndrome.     

A rare case of 46, XX (SRY positive) testicular disorder of sex development with growth hormone deficiency: Case report

Rationale:Chromosome karyotype analysis and SRY (sex determined region of Y chromosome) gene detection are routines for the diagnosis of growth hormone deficiency (GHD), but further whole exome gene sequencing occasionally leads to subversive results and unexpected conclusions.Patient concerns:We report a single case of a 7-year-old Chinese boy who had stunted growth since he was 1 year old. He was short in height (height Standard Deviation Score (SDS) was less than 2.9), bilateral scrotal dysplasia and delayed bone age.Diagnosis:His growth hormone (GH) stimulation tests showed GHD. His karyotype analysis and polymerase chain reaction (PCR) analyses indicated a 46, XX disorder of sex development (DSD) without the presence of the SRY gene. Nevertheless, considering that female gonad was not observed in the chest and abdominal magnetic resonance imaging, the whole exome gene sequencing was performed. Sequencing data confirmed the presence of SRY gene sequence and two copies of chromosome X. Later, using different primer sequences for PCR, it showed that the SRY gene was positive. The final diagnosis was a rare case of “46, XX (SRY positive) testicular DSD with GHD”.Interventions:The boy''s parents agreed to use recombinant human growth hormone (rhGH) for GHD treatment, the starting dose was 0.035 mg / kg / day. But they disagreed with molecular diagnostics and genomic analysis of the Y chromosome.Outcomes:The boy was treated with rhGH for 3 months and his height increased by 2.2 cm. The patient will be followed-up until the end of his puberty.Lessons:In summary, whole exome gene sequencing overturned the preliminary diagnosis results of karyotype analysis and SRY gene detection, and found that there may be a certain correlation between testicular DSD and GHD.     

Pathological gambling in a patient on piribedil: A case report

Rationale:Piribedil is an orally active dopamine agonist that has been widely used for Parkinson disease (PD), with its partial D2/D3 agonistic functions and alpha2-adrenoreceptor antagonistic effects, piribedil has been proved to be efficacious in the relief of motor symptoms in PD, while it can also lead to impulse control disorders such as pathological gambling due to its dopamine agonistic effects.Patient concerns:A 28-year-old Chinese female patient with Parkinson disease and a history of taking piribedil finally developed pathological gambling and depressive episode.Diagnoses:After a careful clinical observation and evaluation, the patient met the criteria of severe depressive episode and pathological gambling due to antiparkinson therapy.Interventions:We discontinued piribedil and picked bupropion, a dopamine reuptake inhibitor, to alleviate the depressive symptom. Benzhexol and selegiline were also added for the control of motor fluctuations.Outcomes:After 3 weeks’ treatment, the patient''s depressive mood was significantly alleviated and her recurring PD symptoms were also relieved. She was no more addicted to network gambling, and there was no recurrence during the 1-year follow-up.Lessons:Piribedil-induced problem gambling and impulse control disorders are side effects needed to be evaluated when commencing a patient on piribedil. This case further emphasizes the importance of monitoring and controlling Parkinson symptoms after drug reduction or withdrawal. Anticipation of this risk strengthens the significance of detailed medical history-taking and targeted clinical management.     

Neuromyelitis optica spectrum disorder with active replication of hepatitis B virus and seropositive anti-aquaporin-4 antibody: A case report

Rationale:Neuromyelitis optica spectrum disorder (NMOSD) associated with active replication of hepatitis B virus (HBV) is rare. High-dose corticosteroids are the mainstay treatment of NMOSD; however, these may cause reactivation of viral replication in patients with stable HBV which may lead to liver damage. Therefore, care should be placed in corticosteroid use in patients with NMOSD and HBV infection.Patient concerns:Herein, we report the case of a 31-year-old woman with NMOSD and HBV infection who was seropositive for anti-aquaporin-4 antibody. The stable and HBV carrier status of the patient led to the deferment of antiviral and hepatoprotective agents in early treatment. However, liver function impairment was detected during follow-up, with an improvement in the best-corrected visual acuity.Diagnoses:The patient was diagnosed with NMOSD with active replication of HBV and seropositive anti-aquaporin-4 antibody considering the medical history and ancillary examinations.Interventions:To manage NMOSD, intravenous high-dose methylprednisolone (20 mg/kg d) was administered for 5 days which was gradually tapered to oral steroids. However, liver function impairment was observed during follow-up; therefore, anti-HBV drugs (entecavir) and hepatoprotective drugs (bicyclol or polyunsaturated phosphatidylcholine) were administered.Outcomes:A marked improvement was observed in the patient''s best-corrected visual acuity after 4 weeks of treatment. However, follow-up examinations revealed liver function damage which necessitated administration of antiviral and hepatoprotective drugs. Liver function normalized after 1 month.Lesson:This case underscores the importance of preventive treatment of liver protection in patients with HBV infection prior to or simultaneous with glucocorticoid therapy and furthermore, there is an urgent need to develop authoritative guidelines regulating corticosteroid use in the treatment of patients with HBV infection.     

Never too late: A commentary on “Impact of age, sex and exercise on brachial and popliteal artery remodelling in humans”

Lysyl oxidase (LOX) is an enzyme critical for the stability of extracellular matrix and also known to have diverse biological functions. Little is known, however, about the role of LOX in regulating inflammation. Here we demonstrate that LOX suppresses secretion of monocyte chemoattractant protein-1 (MCP-1) in cultured vascular smooth muscle cells. Furthermore, enhancement of LOX activity reduces MCP-1 in a mouse model of abdominal aortic aneurysm (AAA), thereby preventing macrophage infiltration and AAA progression. These findings suggest that LOX has a novel function in resolving inflammation by reducing MCP-1 in AAA.     

A trichobezoar in a child with undiagnosed celiac disease: A case report

Celiac disease is a chronic, immune-mediated enteropathy caused by a permanent sensitivity to ingested gluten cereals that develops in genetically susceptible individuals. The classic presentation of celiac disease includes symptoms of malabsorption but has long been associated with cognitive, emotional, and behavioral disorders. We describe an 8-year-old patient with nonscarring alopecia and diagnosed with trichotillomania. Furthermore, she presented with a 3-year history of poor appetite and two or three annual episodes of mushy, fatty stools. Laboratory investigations showed a normal hemoglobin concentration and a low ferritin level. Serologic studies showed an elevated tissue immunoglobulin G anti-tissue transglutaminase level. A duodenal biopsy showed subtotal villous atrophy and crypt hyperplasia, and a large gastric trichobezoar was found in the stomach. Immediately after beginning a gluten-free diet, complete relief of trichotillomania and trichophagia was achieved. In this report, we describe a behavioral disorder as a primary phenomenon of celiac disease, irrespective of nutritional status.     

Reye's syndrome in adults. A case report and review of the literature

Adult Reye's syndrome (ARS) is an infrequently diagnosed condition that typically affects patients younger than age 35 years. We describe a 61-year-old man with ARS occurring after influenza B-USSR infection and aspirin use. The diagnosis of ARS was confirmed by oil-red-O stain of liver biopsy tissue and subsequent electron microscopy. We review the literature on ARS and compare the clinical features and management of ARS with pediatric Rye's syndrome. This case is of interest to practitioners treating adult patients because it demonstrates that the patient population at risk for Reye's syndrome is broader than generally believed.     

Pseudoaneurysm of aortocoronary vein graft secondary to late venous rupture: case report and literature review.

Formation of pseudoaneurysms of saphenous vein grafts after coronary artery bypass grafting has been reported previously in relation to anastomoses or secondary to infection. Pseudoaneurysm of the saphenous vein graft after late rupture of the saphenous vein and containment by the obliterated pericardial cavity has not been documented. Such a case is reported and published reports of similar cases are reviewed.     

Saphenous vein graft ectasia: an unusual late complication of coronary artery bypass surgery. A case report.

Aneurysms and ectasias of saphenous vein grafts are infrequent complications of coronary artery bypass surgery. They usually present as an expanding asymptomatic mediastinal mass on chest x-ray film or computed tomography scan. Though rare, they must be excluded from the differential diagnosis of mediastinal masses to avoid potentially dangerous needle biopsy. The authors describe ectasia of a saphenous vein graft in a 62-year-old man 14 years after coronary artery bypass surgery. The relevant literature is also discussed.     

Tocilizumab in glucocorticoid-resistant graves orbitopathy. A case series report of a mexican population

PurposeTo describe pre- to post-treatment changes in clinical activity score (CAS) and exophthalmometry in patients with Graves orbitopathy treated with tocilizumab (TCZ).Material and methodsEight Mexican patients presenting with active Graves orbitopathy (CAS > 3/7) previously treated with glucocorticoids received 1 monthly dose of TCZ for 6 months. CAS, EUGOGO severity assessment and exophthalmometry were used to evaluate clinical status, with serum measurement of thyroid-stimulating hormone receptor antibodies (TR-Ab) for biochemical evaluation before and after application of TCZ.ResultsEight patients were analyzed: 6 male (75%), 2 female (25%): mean age, 45.9 ± 11.2 years; mean weight, 85 ± 18.3 kg. Mean TR-Ab level at treatment outset was 291.9 ± 96.4%, mean CAS 4.1 ± 0.3 and mean exophthalmometry 21.2 ± 3.2 mm. After TCZ treatment, mean TR-Ab level fell to 172.7 ± 54% (P = 0.001), mean CAS to 1.1 ± 0.6 (P = 0.001) and mean exophthalmometry to 19.3 ± 2 mm (P = 0.02).ConclusionsTCZ is a therapeutic option for glucocorticoid-resistant orbitopathy, and should be considered in second line due to the cost of treatment or in first line in patients with contraindications to intravenous GC pulse therapy.     

Timing of insulin basal rate reduction to reduce hypoglycemia during late post-prandial exercise in adults with type 1 diabetes using insulin pump therapy: A randomized crossover trial

AimsTo compare the efficacy of three timings to decrease basal insulin infusion rate to reduce exercise-induced hypoglycaemia in patients with type 1 diabetes (T1D) using pump therapy.MethodsA single-blinded, randomized, 3-way crossover study in 22 adults that had T1D > 1 year and using insulin pump > 3 months (age, 40 ± 15 years; HbA_1c, 56.3 ± 10.2 mmol/mol). Participants practiced three 45-min exercise sessions (ergocyle) at 60% VO_2peak 3 hours after lunch comparing an 80% reduction of basal insulin applied 40 minutes before (T-40), 20 minutes before (T-20) or at exercise onset (T0).ResultsNo significant difference was observed for percentage of time spent < 4.0 mmol/L (T-40: 16 ± 25%; T-20: 26 ± 27%; T0: 24 ± 29%) (main outcome) and time spent in target range 4.0–10.0 mmol/L (T-40: 63 ± 37%; T-20: 66 ± 25%; T0: 65 ± 31%). With T-40 strategy, although not significant, starting blood glucose (BG) was higher (T-40: 8.6 ± 3.6 mmol/L; T-20: 7.4 ± 2.5 mmol/L ; T0: 7.4 ± 2.7 mmol/L), fewer patients needed extra carbohydrates consumption prior to exercise for BG < 5.0 mmol/L (T-40: n = 3; T-20: n = 5; T0: n = 6) as well as during exercise for BG < 3.3 mmol/L [T-40: n = 6 (27%); T-20: n = 12 (55%); T0: n = 11 (50%)] while time to first hypoglycaemic episode was delayed (T-40: 28 ± 14 min; T-20: 24 ± 10 min; T0: 22 ± 11 min).ConclusionDecreasing basal insulin infusion rate by 80% up to 40 minutes before exercise onset is insufficient to reduce exercise-induced hypoglycaemia.