Osteoporosis in a Woman With Multiple Sclerosis: A Case Report.

Multiple sclerosis is a systemic autoimmune disease characterized by demyelination of nerves within the central nervous system. The prevalence of the disease is increasing. Cases with varying severity are observed. Multiple sclerosis is accompanied by severe osteoporosis, which may lead to fractures and may compromise patient mobility. The aim was to describe the case of a patient with multiple sclerosis who developed severe osteoporosis with multiple fractures. A female patient was diagnosed with multiple sclerosis at the age of 52. At the age of 63, she presented with a fracture of the right femur. She was treated surgically with total arthroplasty. Osteoporosis was diagnosed and treatment was initiated. Seven months later the patient fell upon the fractured leg and developed a periprosthetic femoral fracture. She was treated with open reduction and internal fixation. Thereafter, bisphosphonates were administered. The patient can now walk with difficulty, independently, without orthotic help. In this case report, we have presented a case of multiple sclerosis who developed severe osteoporosis with multiple fractures.

Persistence on subcutaneous tocilizumab as monotherapy or in combination with synthetic disease-modifying anti-rheumatic drugs in rheumatoid arthritis patients in Greece (EMBRACE study): a multicenter, post-marketing, non-interventional, observational trial.

INTRODUCTION: Rheumatoid arthritis (RA) is a systemic, inflammatory disease affecting multiple organs and causing physical disability over time. OBJECTIVE: The primary objective was to evaluate treatment persistence to subcutaneous tocilizumab (TCZ-SC). Additionally, treatment effects on persistence and their associations with clinical and patient-reported outcomes were assessed. METHOD: We performed a multicenter, non-interventional, 52-week observational study on 222 patients with moderate or severe RA. Clinical outcomes were evaluated by using disease activity score for 28 joints (DAS28) and European League Against Rheumatism (EULAR) response, and patients' perceptions were evaluated by using Health Assessment Questionnaire (HAQ), Visual Analog Scale (VAS) for pain, and patient global assessment (PtGA) of disease activity. Safety was assessed throughout the study. RESULTS: The mean age of the overall cohort was 62.2 ± 12.3 years, and 83.8% were females. Persistence to TCZ-SC was 89.6% at week 24 and 85.1% at week 52 in the overall cohort with slightly increased persistence in the combination group. At week 52, changes from the baseline were - 2.68 in DAS28, - 0.76 in HAQ, - 43.21 in VAS pain, and - 41.66 in PtGA (p < 0.0001 for all). Moderate and good EULAR response was achieved in 83.2% of patients. Non-serious and serious adverse events occurred in 18.5% and 3.2% of the participants, respectively. CONCLUSIONS: The current study confirms the favorable safety and effectiveness of TCZ-SC as well as its acceptability by RA patients in Greece, with sustained high persistence rates up to 52 weeks. TCZ-SC offers a sustainable treatment response in RA. Key Points • Based upon clinical and patient-reported outcomes, TCZ-SC is a highly effective and safe treatment modality in patients with moderate-to-severe RA. • Persistence to TCZ-SC was high throughout the study, both as monotherapy and in combination with csDMARDs. • TCZ-SC is effective both as monotherapy and when used in combination with other csDMARDs regardless of the line of treatment.

Vitamin D Levels as a Marker of Severe SARS-CoV-2 Infection.

The SARS-CoV-2 virus may cause severe infection, which is associated with diverse clinical manifestations. Vitamin D has immunomodulating properties and may enhance the body's defense system against invading pathogenic organisms. The aim was to assess 25(OH)D3 levels in patients hospitalized for severe infection from the SARS-CoV-2 virus and explore the relationship between 25(OH)D3 and outcomes. In a group of 88 patients hospitalized for severe infection from the SARS-CoV-2 virus and a control group matched for age and sex, the levels of 25(OH)D3 were analyzed. Levels of 25(OH)D3 were 17.36 ± 8.80 ng/mL (mean ± SD) compared with 24.34 ± 10.34 ng/mL in patients with severe SARS-CoV-2 infection and the control group, respectively, p < 0.001 (Student's t-test). 25(OH)D3 levels were significantly related to outcomes, i.e., survival as opposed to non-survival, as more patients with 25(OH)D3 deficiency (0-10 ng/mL) and insufficiency (10-20 ng/mL) had a fatal outcome as compared with those with vitamin D sufficiency (p < 0.001, chi-square test, p < 0.001, Fisher's exact test). Levels of 25(OH)D3 were inversely related to C-reactive protein (CRP), ferritin, d-dimer, and fibrinogen levels (p < 0.001, linear regression analysis, beta coefficient of variation, -0.176, -0.160, -0.178, and -0.158, respectively). Vitamin D deficiency observed in severe SARS-CoV-2 infection was related to disease outcomes.

Thyroid Disease and Systemic Lupus Erythematosus.

Background and Objectives: Thyroid disease has been associated with autoimmune disorders. As systemic lupus erythematosus (SLE) is a systemic autoimmune disease with diverse manifestations spanning across all organ systems, the relationship of SLE with thyroid disorders needs investigation. In particular, the relationship of SLE with autoimmune thyroid disease has attracted the interest of the research community. The aim was to evaluate the relationship of SLE with autoimmune thyroid disease. Materials and Methods: A cohort of 45 consecutive patients with a mean age of 47.97 years (range 21-79 years) and 45 age- and sex-matched controls were prospectively studied over a period of 12 months for the presence of thyroid disease and the prevalence of antithyroid antibodies. Results: Four patients (8.9%) were found to suffer from primary hypothyroidism, five (11.11%) from subclinical hypothyroidism and one (2.22%) from hyperthyroidism, whereas one (2.22%) of the controls had primary hypothyroidism and one (2.22%) had hyperthyroidism. Five patients (11.11%) had a thyroid hormone profile that was compatible with the presence of euthyroid sick syndrome. Thyroid peroxidase (TPOab) and thyroglobulin (Tgab) antibodies were detected in 20/45 and 15/45 of the SLE population and in 7/45 and 5/45 of the controls, respectively (p < 0.05, chi-square test). Conclusions: In conclusion, the incidence of clinical thyroid disease is greater amongst SLE patients than in a control population, and in a significant number of these patients, antithyroid antibodies are detectable. Thus, a subset of lupus patients appears to be predisposed to the development of thyroid disease, and this should be considered when evaluating patients with SLE.

Current Treatment Approach, Emerging Therapies and New Horizons in Systemic Lupus Erythematosus.

Systemic lupus erythematosus (SLE), the prototype of systemic autoimmune diseases is characterized by extreme heterogeneity with a variable clinical course. Renal involvement may be observed and affects the outcome. Hydroxychloroquine should be administered to every lupus patient irrespective of organ involvement. Conventional immunosuppressive therapy includes corticosteroids, methotrexate, cyclophosphamide, mycophenolate mofetil, azathioprine, cyclosporine and tacrolimus. However, despite conventional immunosuppressive treatment, flares occur and broad immunosuppression is accompanied by multiple side effects. Flare occurrence, target organ involvement, side effects of broad immunosuppression and increased knowledge of the pathogenetic mechanisms involved in SLE pathogenesis as well as the availability of biologic agents has led to the application of biologic agents in SLE management. Biologic agents targeting various pathogenetic paths have been applied. B cell targeting agents have been used successfully. Belimumab, a B cell targeting agent, has been approved for the treatment of SLE. Rituximab, an anti-CD20 targeting agent is also used in SLE. Anifrolumab, an interferon I receptor-targeting agent has beneficial effects on SLE. In conclusion, biologic treatment is applied in SLE and should be further evaluated with the aim of a good treatment response and a significant improvement in quality of life.

Real-world effectiveness of golimumab in adult patients with rheumatoid arthritis, psoriatic arthritis, and axial spondyloarthritis and an inadequate response to initial TNFi therapy in Greece: the GO-BEYOND prospective, observational study.

The impact of golimumab (GLM) on remission or low disease activity (LDA) was evaluated in patients with moderate-to-severe rheumatoid arthritis (RA), progressive psoriatic arthritis (PsA), or severe axial spondyloarthritis (axSpA), who failed previous treatment for their rheumatic disease with one initial tumor necrosis factor α inhibitor (TNFi). This is a multicenter, prospective, real-world observational 18-month study, conducted in Greece. The primary endpoint, assessed at 6 months, included the proportion of patients attaining LDA and/or remission (Disease Activity Score for 28 joints based on C-reactive protein [DAS28-CRP] ≤ 3.2), minimal disease activity (MDA; MDA criteria), and moderate disease activity (Bath Ankylosing Spondylitis Disease Activity Index [BASDAI] score 4-7), respectively. Other endpoints evaluated the persistence to GLM treatment and its impact on patients' work productivity (Work Productivity and Activity Impairment [WPAI] instrument) and quality of life (QoL; EuroQoL5 dimensions 3 levels [EQ-5D-3L] questionnaire). Descriptive statistics, the Wilcoxon signed-rank test, and Kaplan-Meier method were used for analyses. At 6 months, LDA was achieved by 46.4% of patients with RA, MDA by 57.1% of patients with PsA, and BASDAI 4-7 by 24.1% of patients with axSpA. For all study patients, persistence rates on GLM were high (85.1-93.7%) over 18 months; all WPAI domain scores and the EQ-5D-3L index score improved significantly (p < 0.001) from baseline to 18 months. GLM treatment was effective in patients with RA, PsA, or axSpA who had failed previous treatment with one TNFi and led to significant WPAI and QoL improvements. Persistence rates were high. Trial registration number and date of registration: As per the local regulations the study has been registered at the national registry for non-interventional studies https://www.dilon.sfee.gr/studiesp_d.php?meleti_id=MK8259-6995 .

Sarcopenia in patients with diabetes mellitus.

INTRODUCTION: Diseases such as diabetes mellitus may be associated with adverse changes in body composition. Sarcopenia is characterized by a progressive and generalized loss of skeletal muscle mass and functionality. AIM: To investigate the relationship between type 2 diabetes mellitus (T2DM) and sarcopenia. MATERIALS AND METHODS: In a retrospective, non-randomized study, 35 T2DM patients, aged 20-80 years, were assessed for sarcopenia prevalence compared to controls (n=16). Appendicular skeletal mass (ASM) (kg) was measured, and sarcopenia was defined as SMI <7.0 and <5.7 kg/m2, in males and females, respectively, using the European Working Group on Sarcopenia in Elderly (EWGOSP) definition. Low physical performance was defined as a walking speed of <0.8 m/s. RESULTS: Incidence of sarcopenia was significantly higher in T2DM patients vs. controls (27% vs. 20%, p=0.01) and elderly vs. young participants (40% vs. 12%, p<0.001), respectively. Walking velocity was significantly lower in T2DM patients compared to male and female controls (1.08±0.22 vs. 1.23±0.18 and 1.07±0.26 vs. 1.26±0.16, respectively, p<0.001,). CONCLUSIONS: A moderate prevalence of sarcopenia in patients with type 2 diabetes mellitus was observed, which appeared to increase significantly in older men. Finally, incidence of T2DM displayed decreased physical performance in both genders.

Vitamin D Deficiency in Primary Sjögren's Syndrome: Association with Clinical Manifestations and Immune Activation Markers.

Vitamin D is an agent involved in bone and mineral homeostasis. It has been recognized as a potent immunomodulator. It has immune-enhancing properties, and it induces immune tolerance. Vitamin D deficiency has been shown to be related to the development of autoimmune disorders. Vitamin D deficiency has been observed in patients with rheumatoid arthritis (RA) and it has been shown to be related with disease activity. Vitamin D deficiency has also been found in patients with systemic lupus erythematosus (SLE) and it was shown to be related to disease activity and renal involvement. Vitamin D deficiency has also been observed in multiple sclerosis. Vitamin D has been found to act as a supplemental therapeutic agent in multiple sclerosis. Sjögren's syndrome is a systemic autoimmune disease affecting the exocrine glands, known as an autoimmune epithelitis. The disease has a complex pathogenesis, requiring a genetic background, immune cell activation, and autoantibody production. The disease affects the exocrine glands, lacrimal, and salivary glands leading to ocular and oral dryness. Vitamin D levels have been measured in patients with Sjögren's syndrome and an association was observed between low vitamin D levels, peripheral neuropathy and the presence of lymphoma. In other cohorts, such as a Turkish cohort, vitamin D deficiency was observed in patients with Sjögren's syndrome. The aim is to measure serum vitamin D levels in consecutive patients with primary Sjögren's syndrome and investigate the relationship between vitamin D levels and the presence of immunologic markers, clinical, serological, and histopathological characteristics.

Vitamin D levels in Greek patients with systemic lupus erythematosus.

OBJECTIVES: Vitamin D deficiency has been observed in autoimmune rheumatic diseases, such as rheumatoid arthritis. The aim was to study vitamin D in patients with systemic lupus erythematosus (SLE) and its relationship with disease activity. METHODS: In a cohort of 45 patients with SLE, 41 females and 4 males, aged 47.07 ± 2.17 years (mean ± SEM), and range = 21-79 years, 25(OH)D3 levels were determined by electrochemiluminescence. C3 and C4 levels were also analyzed. SLE disease activity was estimated by SLEDAI-2K. Observations were also performed in a control group matched for age and sex. RESULTS: In this cohort of SLE patients, 25(OH)D3 levels were 40.36 ± 2.41 nmol/L (mean ± SEM) as opposed to 60.98 ± 4.28 nmol/L in the control group (p < 0.001, Student's t test). Vitamin D levels were related to C3 (p < 0.001, linear regression analysis), correlation coefficient 0.106, r2 = 0.011, and C4 (p < 0.001); correlation coefficient 0.316 and r2 = 0.100; and inversely related to disease activity (p < 0.001), correlation coefficient -0.572 and r2 = 0.327. 25(OH)D3 levels were 17.73 ± 1.20 nmol/L and 12.24 ± 0.93 nmol/L, in the groups without and with renal involvement, respectively (p = 0.001, Student's t test). CONCLUSIONS: Vitamin D levels are low in SLE patients and are inversely related to disease activity. Routine screening for vitamin D levels should be performed in SLE patients.

The effects of golimumab on work productivity and quality of life among work-active axial spondyloarthritis and psoriatic arthritis patients treated in the routine care in Greece: the 'GO-UP' study.

PURPOSE: To examine the impact of golimumab, on work productivity, activity limitation, and quality of life (QoL) in patients with axial spondyloarthritis (axSpA) and psoriatic arthritis (PsA). METHODS: This real-world, multicenter, prospective study consecutively enrolled adult consented work-active patients with axSpA or PsA, newly initiated on golimumab as per the approved label. Prior receipt of > 1 prior biologic, or switching from another tumor-necrosis factor inhibitor due to primary non-response or safety reasons was not allowed. The Work Productivity and Activity Impairment-Specific Health Problem and the EuroQol 5-Dimensions (EQ-5D)-5-Level instruments were completed by the patients to assess the impact of golimumab on work productivity and activity impairment, and generic QoL, respectively. RESULTS: Overall, 121 eligible patients (mean age: 45.4 years; median disease duration: 11.3 months), 51 diagnosed with PsA and 70 with axSpA, were enrolled by 19 rheumatologists. Over a 11.9-month median observation period, < 1% of injections were missed (as collected by patient diaries), and the 12-month golimumab retention rate was 91.7%. At 3, 6, and 12 months post baseline, in the overall population, work productivity loss improved by a median of 31.4%, 44.2%, and 50.0%; activity impairment improved by 40.0%, 40.0%, and 50.0%; and the EQ-5D UK-weighted utility index improved by 0.24, 0.32, and 0.36 points, respectively (p < 0.001 for all). Statistically significant improvements in these measures were also noted in the PsA and axSpA subpopulations. CONCLUSION: In the routine care in Greece, golimumab displays beneficial effects on work productivity, daily activities, and QoL in work-active patients with axSpA and PsA. TRIAL REGISTRATION: Trial registration number and date of registration: As per the local regulations the study has been registered at the national registry for non-interventional studies https://www.dilon.sfee.gr/studiesp_d.php?meleti_id=MK8259-6083 .

Nutritional Pearls: Diet and Rheumatoid Arthritis.

Various studies have investigated the effect of diet on rheumatoid arthritis (RA) as a complimentary treatment along with standard drug therapy. Various types of diet have been investigated. Fasting, the Mediterranean diet, the Cretan Mediterranean diet, vegetarian diet, an anti-inflammatory diet and the use of various specific food substances have been in the focus of research interest. The relationship of obesity with disease activity in RA has also been investigated. A period of fasting followed by Mediterranean diet, the Cretan Mediterranean diet and an anti-inflammatory diet have been found to have a beneficial effect on disease activity in RA. Obesity has been found to be associated with increased disease activity in RA. However, weight loss appears to be related to increased mortality in RA. The use of flavonoids, resveratrol and curcumin may have a beneficial effect in RA. It appears that diet may aid in RA management as a complimentary factor to standard drug treatment.

The Effect of Omega-3 Fatty Acids on Rheumatoid Arthritis.

Omega-3 fatty acids are unsaturated fatty acids thought to play a role in health and disease. They are known as essential fatty acids, as they cannot be synthesized in mammals. Omega-3 fatty acids have a beneficial effect on the secondary prevention of coronary artery disease and stroke and are essential for the development and function of the nervous system and the retina in man. Omega-3 fatty acids are thought to have immunomodulatory properties as they act as precursors to lipid mediators of inflammation which may limit or modulate the inflammatory response. Omega-3 fatty acids seem to prevent or attenuate experimental arthritis. They may have a beneficial effect in the treatment of rheumatoid arthritis. Clinical studies have shown that omega-3 fatty acids may have a modulatory effect on disease activity, namely on the number of swollen and tender joints. It appears that omega-3 fatty acids may modulate disease activity in rheumatoid arthritis.

Correlation of patient preferences to treatment outcomes in patients with rheumatoid arthritis treated with tumour necrosis factor inhibitors in Greece.

OBJECTIVES: To investigate possible associations between rheumatoid arthritis (RA) patient-expressed preferences over anti-tumour necrosis factor (anti-TNF) treatment and clinical and patient-reported outcomes. METHODS: PANORAMA was a non-interventional, prospective, multicentre, cohort study of 12 months duration, in patients with moderate-to-severe RA who initiated or switched to anti-TNF treatment. After initiation of anti-TNF, patients completed a preferences questionnaire on attributes related to anti-TNF treatment. Satisfaction with treatment was assessed with the Treatment Satisfaction Questionnaire for Medication (TSQM); compliance and persistence to treatment were recorded via a patient diary. Univariate and multivariate analyses were applied to assess correlations between patients' preferences over treatment with clinical and patient-reported outcomes. RESULTS: A total of 254 patients were enrolled; 66.1% (168/254) had highly active disease (DAS28-ESR > 5.1), while 65.4% (166/254) were biological-naïve. The 12-month drug-survival rate was 72.3%, while the respective rates of good EULAR response and remission (DAS28-ESR < 2.6) were 56.5% and 40.8%, respectively. By univariate analysis, fulfilment of patient preferences over treatment was associated with increased probability of remaining on therapy (p = 0.019), good EULAR response (p < 0.001) and satisfaction with treatment (p < 0.001). By multivariate analysis, fulfilment of patient preferences was the most important predictor for good EULAR response (OR 5.56, p < 0.001; finding confirmed and after propensity scoring matching), while seropositivity (HR 1.18, p = 0.047) and a high ESR (> 35 mm/h, HR 1.16, p = 0.071) predicted drug survival. CONCLUSIONS: In anti-TNF-treated RA patients, fulfilment of treatment preferences was independently associated with a good EULAR response and correlated with drug persistence at 12 months, emphasising the importance of patient preferences in treatment outcomes. Key Points • In anti-TNF treated RA patients, fulfilment of patients' treatment preferences is associated with a good clinical response at 12 months. • A shared decision-making process can maximise treatment's outcome in anti-TNF treated patients.

Vitamin D in acutely ill patients.

Objective To investigate 25(OH)D3 levels and their relationship to survival in a cohort of acutely ill patients on admission to an intensive care unit. Methods This study enrolled acutely ill patients at admission to an intensive care unit and a group of sex- and age-matched healthy control subjects. The 25(OH)D3 levels were measured using an enzyme immunoassay. C-reactive protein and procalcitonin levels were also measured using immunoassays. Results A total of 50 acutely ill patients and 50 healthy control subjects were enrolled in the study. The mean ± SEM 25(OH)D3 levels were significantly lower in the acutely ill patients compared with the control group (11.74 ± 0.88 ng/ml versus 24.66 ± 1.60 ng/ml, respectively). The 25(OH)D3 levels were not related to survival. An inverse relationship was observed between 25(OH)D3 levels and C-reactive protein levels. A weak inverse relationship was also observed between 25(OH)D3 levels and procalcitonin levels. Conclusions The 25(OH)D3 levels were decreased in acutely ill patients admitted to an intensive care unit compared with healthy control subjects. 25(OH)D3 levels may be inversely related to C-reactive protein and procalcitonin levels.

Orf disease in a patient with rheumatoid arthritis.

Orf disease is a viral infection, affecting patients being involved in the care of livestock either professionally or habitually. It is also known as ecthyma contagious, contagious pustular dermatitis or infectious euphoria being a rare zoonotic disease caused by an epitheliotropic DNA virus from the parapoxvirus group. We report a case of Orf disease affecting the hand of a patient with rheumatoid arthritis (RA) on treatment with methotrexate and adalimumab, an anti-tumor necrosis factor biological agent. The patient was successfully treated with doxycycline, while immunosuppressive treatment was discontinued.

Anti-synthetase syndrome presenting with interstitial lung disease.

Anti-synthetase syndrome is an idiopathic inflammatory myopathy characterized by interstitial lung involvement, arthritis, Raynaud's phenomenon, mechanic's hands and fever. The case of anti-synthetase syndrome in a young female is reported. The patient presented with interstitial lung disease, initially subclinical muscle involvement, Raynaud's phenomenon, arthritis and mechanic's hands. Induction therapy was administrated, that consisted of intravenous methylprednisolone and cyclophosphamide, followed by azathioprine and prednisone with good response, and finally, complete remission. The presence of mechanic's hands in a patient with lung involvement should prompt for specific investigations such as anti-synthetase antibodies and muscle enzymes. Anti-synthetase syndrome should be aggressively managed by immunosuppressive therapy, as interstitial lung disease is a severe manifestation of the disease which may affect quality of life and life expectancy.

Safety profile of repeated rituximab cycles in unselected rheumatoid arthritis patients: a long-term, prospective real-life study.

OBJECTIVES: To evaluate the long-term safety of rituximab (RTX) in rheumatoid arthritis (RA) patients in daily clinical practice. METHODS: This was a multicentre (17 Greek Rheumatology sites), prospective, long-term, pharmacovigilance study of patients with moderate to severe RA and an inadequate response or intolerance to ≥1 anti-tumour necrosis factor (TNF) agents. Adverse events (AEs) were recorded and collected prospectively every 2-6 months. RESULTS: 234 patients (mean age: 59±12.5, 79.5% women, mean DAS28: 5.35±1.32) were included and followed for 27.7 months (median). The overall AEs, serious AE (SAEs) and serious infection (SIEs) rate were 48.36, 6.68 and 2.53/100 patient-years, respectively. Three cases of hepatitis B virus (HBV) reactivation were recorded (two in chronic and one in past HBV infection). Withdrawals due to AEs (5.6%) occurred more frequently during the first cycles of RTX therapy while repeated RTX cycles were not associated with an increased risk of AEs. There were 3 deaths with an incidence rate of 0.69/100 patient-years. Age ≥65 years was associated with a higher incidence rate ratio of AEs and SAEs as compared to <65 years (1.53, p=0.002 and 2.88, p=0.005, respectively). Drug retention rate during 434.28 patient-years of follow-up was 57.3%. Factors associated with drug discontinuation by multivariate analysis included age, baseline swollen joint count and no use of concomitant methotrexate therapy. CONCLUSIONS: Long-term RTX therapy in a real-life RA cohort, did not reveal any new safety issues. Advanced age was associated with increased risk of AEs and premature drug discontinuation.

Vitamin D deficiency in a patient with HDR syndrome.

The case of a patient with clinical symptoms, laboratory and imaging findings of hypoparathyroidism, sensorineural deafness, renal dysplasia HDR, or Barakat syndrome (hypoparathyroidism, deafness, renal dysplasia), and vitamin D deficiency, is presented. A Caucasian man aged 51 years with a history of chronic hypocalcaemia since childhood, was admitted with hypertonia of the body and extremities, and loss of consciousness. On admission, he was found to have severe hypocalcaemia, hyperphosphataemia, severe hypoparathyroidism, low serum magnesium and mild renal insufficiency. Calcium gluconate was administered intravenously supplemented with magnesium, and the patient recovered consciousness while clinical and laboratory findings improved. Evaluation revealed left renal aplasia and sensorineural deafness affecting both ears. Vitamin D deficiency was also present. He was given calcium and vitamin D supplements orally, and the hypocalcaemia was corrected. This case is described as it is an extremely rare case of HDR syndrome with concurrent vitamin D deficiency.