scholarly journals A new therapeutic approach with tocilizumab in a 39-year-old patient with recurrent diabetic myonecrosis

Author(s):  
Judith Wagemann ◽  
Sarah Keller ◽  
Maria de las Mercedes Noriega ◽  
Werner Stenzel ◽  
Udo Schneider ◽  
...  

ABSTRACT We report the case of a 39-year-old female patient with acute painful swelling of the left thigh and symmetric muscle weakness in both upper legs. The patient had a history of long-standing, poorly controlled type 1 diabetes which required dialysis. Serum inflammatory markers were highly elevated. Magnetic resonance imaging (MRI) indicated necrotic or inflammatory colliquation. As antibiotic therapy did not lead to clinical improvement, a successful anti-inflammatory therapy with prednisolone was initiated. Three months later, the patient presented with a new onset of progressive and painful muscle swelling of the right thigh. MRI showed pronounced swelling of the right adductor muscles and inflammatory markers were massively elevated. In the absence of autoantibodies or any infectious agents and the recurrent symptomatology, relapsing diabetogenic myonecrosis was diagnosed. Initially, clinical improvement could only be achieved with high-dose glucocorticosteroids. Intravenous immunoglobulins did not show an effect, whereas serological and clinical remission was achieved after we administered tocilizumab intravenously. Diabetic myonecrosis is a rare complication of long-term, poorly controlled diabetes mellitus. Acute muscle pain and elevated inflammatory markers should prompt suspicion. Contralateral muscle involvement is also suggestive of the disease. The optimisation of diabetes treatment is crucial in order to prevent further disease complications.

2020 ◽  
Vol 48 (7) ◽  
pp. 030006052093211
Author(s):  
Yu Cui ◽  
Zhong-Xi Yang ◽  
Chun-Mei Wang ◽  
Zhan-Peng Zhu

Microvascular decompression (MVD) is an effective and safe approach for treating hemifacial spasm (HFS). Postoperative complications may include facial nerve palsy, hearing loss, intracerebral haematoma, and brainstem infarction. The occurrence of intracranial cyst following MVD is extremely rare, with few cases documented in the literature. Herein, the cases of two patients with HFS who developed ipsilateral cerebellar cyst following MVD are reported. The first patient was a 50-year-old male presenting with a 6-year history of HFS on the right side of his face. MVD was performed, and 12 days postoperatively he developed dizziness and nausea. Magnetic resonance imaging (MRI) showed a cyst in the ipsilateral cerebellum. Antibiotic treatment provided no benefit, and the cyst was drained. The second patient was a 44-year-old female presenting with a 4-year history of HFS on the right side of her face. MVD was performed, and 18 days following surgery, she developed dizziness and nausea. MRI showed an ipsilateral cerebellar cyst. Conservative treatment was applied and the cyst shrunk. At the 2-month follow-up appointment, symptoms were completely resolved in both patients. Cerebellar cyst is a rare complication following MVD. Timely diagnosis and appropriate treatment should be emphasized, and surgical treatment may be unnecessary.


Author(s):  
Rita Ribeiro Dias ◽  
Inês Henriques Ferreira ◽  
Raquel Faria

We present a case of a 56-year-old man with a history of episcleritis (left) and cluster headache (left) who had a penetrating trauma of the left eye leading to amaurosis 1 month previously. Since then, he developed multiple cranial neuropathy of the right side (V, VII, VIII, IX, X, XI and XII cranial pairs). Magnetic resonance imaging (MRI) revealed an infiltrative lesion of the base of the skull which extended to the retropharyngeal and jugular space, which progressed to multiple leptomeningeal masses extending to the clivus, despite aggressive immunosuppression. Rebiopsy of 1 meningeal mass supported the diagnosis of neurosarcoidosis. The patient finally responded to high-dose prolonged infliximab therapy, with complete remission.


2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A377-A378
Author(s):  
Gayane Tumyan ◽  
Aaron Oubre ◽  
Mark Feldman

Abstract Background: Diabetic myonecrosis is a rare complication of long-standing, poorly controlled diabetes mellitus, and is more common in patients with microvascular complications. The diagnosis is frequently missed due to symptoms mimicking other conditions associated with diabetes mellitus. Clinical Case: A 31-year-old woman with a past medical history of long-standing type I diabetes mellitus, end-stage renal disease, diabetic retinopathy, and cataracts presented to our hospital with a chief complaint of right thigh pain and swelling for a week. She did not have any other relevant symptoms and denied a history of trauma. On admission, physical examination revealed right thigh edema, induration, tenderness on palpation, more pronounced in the medial aspect of the thigh, and decreased range of motion of the right hip. Laboratory analysis showed leukocytosis of 13.29 k/uL (normal = 4.23 - 9.71 k/uL) with neutrophilic predominance of 91.4 %, elevated inflammatory markers (ESR > 130 mm/h (normal = 0 - 30 mm/h), CRP 33.84 mg/dL (normal <= 0.49 mg/dL)) and elevated CK levels of 1675 U/L (normal = 29 - 168 U/L). Additionally, the patient was anemic with a hemoglobin level of 8.4 g/dL (normal = 11.0 - 15.0 g/dL) and had a creatinine level of 2.67 mg/dL (normal = 0.55 - 1.11 mg/dL). Hemoglobin A1C level was 10.8 % (normal = 4.0 - 6.0 %). Blood cultures were drawn and did not grow any organisms. A doppler ultrasound of the right leg was negative for a deep venous thrombosis. CT angiogram of right lower extremity revealed diffuse calcifications in the walls of small and medium vessels and edematous changes in thigh musculature associated with subcutaneous edema. Noncontrast MRI revealed localized edema within the vastus medialis, sartorius, and right thigh adductor muscles. The patient improved with analgesics, rest, and gentle physical therapy. She was counseled on compliance with insulin and advised for a close follow up with her endocrinologist. Conclusion: This case reinforces the importance of including diabetic myonecrosis in the differential diagnosis of acute to subacute nontraumatic muscular pain in diabetic patients, particularly in patients with poor glycemic control and known complications. While muscle biopsy can be performed for histological confirmation, the typical imaging findings and clinical presentation can lead to the diagnosis, making further invasive testing unnecessary. In most described cases, the course of this condition is self-limiting.


2009 ◽  
Vol 29 (02) ◽  
pp. 155-157 ◽  
Author(s):  
H. Hauch ◽  
J. Rischewski ◽  
U. Kordes ◽  
J. Schneppenheim ◽  
R. Schneppenheim ◽  
...  

SummaryInhibitor development is a rare but serious event in hemophilia B patients. Management is hampered by the frequent occurrence of allergic reactions to factor IX, low success rates of current inhibitor elimination protocols and the risk of development of nephrotic syndrome. Single cases of immune tolerance induction (ITI) including immunosuppressive agents like mycophenolat mofetil (MMF) or rituximab have been reported. We present a case of successful inhibitor elimination with a combined immune-modulating therapy and high-dose factor IX (FIX). This boy had developed a FIX inhibitor at the age of 5 years and had a history of allergic reactions to FIX and to FEIBA→. Under on-demand treatment with recombinant activated FVII the inhibitor became undetectable but the boy suffered from multiple joint and muscle bleeds. At the age of 11.5 years ITI was attempted with a combination of rituximab, MMF, dexamethasone, intravenous immunoglobulins and high-dose FIX. The inhibitor did not reappear and FIX half-life normalized. No allergic reaction, no signs of nephrotic syndrome and no serious infections were observed.


2019 ◽  
pp. 1-3
Author(s):  
Bertrand Ng ◽  
Arafat Yasser

Omental infarct is a rare cause of an acute abdomen that arises from an interruption of blood supply to the omentum. Here, we present a case of omental infarct in a 67-year-old gentleman with background history of diabetes mellitus who present unusually with a severe acute onset right hypochondrium pain. Examination revealed that he was tender to touch at the right and was having localized guarding. His inflammatory markers were normal. He was successfully treated with laparoscopy surgery and he was subsequently discharged the following day. Omental infarct cases with right hypochondrium pain can sometimes mimicked acute cholecystitis and management includes laparoscopic surgery which can hasten symptoms resolution and reduces hospital stay, however recommendation for surgery has to be balanced with anesthetics risk and complication of the surgery itself.


2020 ◽  
Vol 13 (12) ◽  
pp. e237076
Author(s):  
George Vatidis ◽  
Eirini I Rigopoulou ◽  
Konstantinos Tepetes ◽  
George N Dalekos

Hepatic brucelloma (HB), a rare manifestation of brucellosis, refers to liver involvement in the form of abscess. A 35-year-old woman stockbreeder was admitted due to 1-month history of evening fever, sweating and weight loss, while she was on 3-week course of rifampicin/doxycycline for suspected brucellosis. On admission, she had hepatosplenomegaly and a systolic murmur, while cholestasis, increased inflammation markers and a strong-positive Wright-Coombs test were the main laboratory findings. As blood and bone marrow cultures were unrevealing, further investigation with CT imaging showed a central liver calcification surrounded by heterogeneous hypodense area being compatible with HB. Material from CT-guided drainage tested negative for Brucella spp. After failure to improve on a 10-week triple regiment, surgical excision was decided and Brucella spp were identified by PCR. Our case highlights challenges in establishing HB diagnosis, which should be considered on the right epidemiological context and when serological and radiological evidence favour its diagnosis.


2021 ◽  
Vol 49 (5) ◽  
pp. 030006052110037
Author(s):  
Liu Liang ◽  
Wang Jiajia ◽  
Li Shoubin ◽  
Qi Yufeng ◽  
Wang Gang ◽  
...  

We report the disease characteristics, diagnosis, and treatment of granulomatous orchitis. A 38-year-old man presented with a history of intermittent swelling, pain, and discomfort in the right testicle of 3 days’ duration. Unenhanced magnetic resonance imaging (MRI) of the testis and scrotum revealed an oval mass in the right testis measuring approximately 17 mm in diameter, with clear borders and a target ring-like appearance from periphery to center. T1-weighted imaging (T1WI) showed uniform low-intensity signals, and T2WI showed mixed high- and low-intensity signals. Diffusion-weighted imaging (DWI) signals were iso-intense, and the outer ring on enhanced scans showed progressive enhancement. We performed radical resection of the right testis under combined spinal–epidural anesthesia. The pathological diagnosis was granulomatous right orchitis. Two months postoperatively, ultrasonography showed no testis and epididymal echo signals in the right scrotum, and no obvious abnormalities; color Doppler blood flow imaging (CDFI) findings were normal. Granulomatous orchitis is rare in clinical practice, and the cause is unknown. The disease involves non-specific inflammation; however, it is currently believed that antibiotics and steroids are ineffective for conservative treatment, and orchiectomy should be actively performed.


CNS Spectrums ◽  
2010 ◽  
Vol 15 (S4) ◽  
pp. 3-6 ◽  
Author(s):  
Andres M. Kanner ◽  
Andrew J. Cole

A 27-year-old woman presented to the emergency room after having witnessed generalized tonic clonic seizure while asleep. Birth and development were normal. She had suffered a single febrile seizure at 13 months of age, but had no other seizure risk factors. She was otherwise well except for a history of depression for which she was taking sertraline. Depressive symptoms had been well controlled over the past 3 months, but she had been under increased stress working to finish a doctoral thesis. Neurological examination was normal. Magnetic resonance imaging (MRI) showed modest asymmetry of the hippocampi, slightly smaller on the right, but no abnormal signal and well-preserved laminar anatomy. An electroencephalogram was negative. She was discharged from the emergency room with no treatment. Three weeks later, the patient's boyfriend witnessed an episode of behavioral arrest with lip smacking and swallowing automatisms lasting 45 seconds, after which the patient was confused for 20–30 minutes. The next morning she and her boyfriend kept a previously scheduled appointment with a neurologist.


2019 ◽  
Vol 12 (3) ◽  
pp. e228204
Author(s):  
Frances Varian ◽  
Harpreet Kaur ◽  
Stuart Carter ◽  
Julian Gunn

We present a case of constrictive pericarditis with concomitant blood and bone marrow appearances of chronic myelomonocytic leukaemia (CMML). Despite surgical treatment with pericardiectomy, the patient deteriorated into multiorgan failure. Pericardial histology disclosed a typical inflammatory picture with no evidence of monocytic or malignant infiltrate. Following intensive collaboration between cardiologists, haematologists and rheumatologists via daily email exchanges, a diagnosis was reached of autoinflammatory constrictive pericarditis with a non-infiltrative coexisting CMML. The key to achieving a rapid and sustained response was a trial of high-dose steroids followed by intravenous immunoglobulins. This achieved restoration of cardiac function, resolution of symptoms and near normalisation of inflammatory markers. A diagnosis of concurrent CMML was confirmed at 3 months. The patient remains well, taking colchicine and steroids.


2019 ◽  
Vol 14 (3) ◽  
pp. 246-248 ◽  
Author(s):  
Dhruvkumar M. Patel ◽  
Mukundkumar V. Patel ◽  
Akash D. Patel ◽  
Jignesh C. Kaklotar ◽  
Greshaben R. Patel ◽  
...  

Background:Calciphylaxis is a complex dermatological lesion of micro vascular calcification that is typically presented as panniculitis with gangrenous painful lesions having uremic and non-uremic causes.Case Report:We present a case of a 48-year old male with a history of paroxysmal atrial fibrillation and hypertension taking amlodipine 5 mg and warfarin 5 mg daily for the last 26 months. The patient had a 6- months history of painful swelling followed by necrotic skin ulcer over the right leg. His remarkable examination findings were right leg tender ulcer with surrounding erythema and secondary sepsis. His hemogram, metabolic profile and connective tissue diseases work up were unremarkable except leucocytosis and raised inflammatory markers. His local part radiological and skin biopsy findings were suggestive of calciphylaxis.Results and Conclusion:In our case, warfarin and amlodipine were culprit drugs for the lesion, but Naranjo score (warfarin 7and amlodipine 1) speculate warfarin as a probable adverse reaction of warfarin. The lesion was cured with local wound treatment after discontinuation of warfarin. The physician should be aware of this rare cutaneous disorder of systemic origin for proper management.


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