Pyrexia, pelvic pain and thrombocytopenia in an Asian man with Paget's disease of bone

SIR, We report the case history of a Bangladeshi man with Paget'sdisease of bone who presented with pelvic pain, thrombocytopeniaand pyrexia and was found to have developed angiosarcoma. A 58-yr-old Bangladeshi man was admitted with a 4-week historyof bilateral buttock pain. There was no history of precedingtrauma. Six months previously, he was noted to have thrombocytopeniaand following bone marrow aspiration and biopsy, immune thrombocytopeniawas diagnosed. His antiplatelet antibodies, however, were negative.Physical examination showed a temperature of 36.8°C.     

Pain in chronic pancreatitis: the role of reorganization in the central nervous system

BACKGROUND & AIMS: In various chronic pain conditions cortical reorganization seems to play a role in the manifestations. The aim of this study was to investigate cortical reorganization in patients with pain caused by chronic pancreatitis. METHODS: Twelve healthy subjects and 10 patients with chronic pancreatitis were included. The esophagus, stomach, and duodenum were stimulated electrically at the pain threshold using a nasal endoscope. The electroencephalogram was recorded from 64 surface electrodes and event-related brain potentials (EPs) were obtained. RESULTS: As compared with healthy subjects, the patient group showed decreased latencies of the early EP components (N1, P < .001; P1, P = .02), which is thought to reflect the exogenous brain pain processing specifically. Source analysis showed that the dipolar activities corresponding to the early EPs were located consistently in the bilateral insula, in the anterior cingulate gyrus, and in the bilateral secondary somatosensory area. The bilateral insular dipoles were localized more medial in the patient group than in the healthy subjects after stimulation of all 3 gut segments (P < .01). There also were changes in the cingulate cortex where the neuronal source was more posterior in patients than in controls to stimulation of the esophagus (P < .05). CONCLUSIONS: The findings indicate that pain in chronic pancreatitis leads to changes in cortical projections of the nociceptive system. Such findings also have been described in somatic pain disorders, among them neuropathic pain. Taken together with the clinical data this suggests a neuropathic component in pancreatic pain, which may influence the approach to treatment.     

Bilateral pulmonary infiltrates in a patient with ulcerative colitis receiving mesalazine

A 36-year-old woman presented with a 2-month history of dry cough, bilateral pain in the upper chest, and low-grade fever. She had a 1-year history of ulcerative colitis (UC), which was treated with mesalazine. Cultures of sputum and bronchoalveolar lavage (BAL) fluid were negative. Chest radiograph and a computed tomography (CT) scan showed dense bilateral subpleural infiltrates in both upper lobes. A c-ANCA test was positive in a 1:1280 titer, and further specification showed antibodies against proteinase-3 antigen. Due to the possibility of mesalazine toxicity, this medication was stopped. Within 2 weeks, the patient's symptoms markedly improved, together with the chest roentgenogram.     

Regional cerebral blood flow during gastric balloon distention in functional dyspepsia

BACKGROUND & AIMS: Hypersensitivity to proximal gastric distention as a result of abnormal central nervous system processing of visceral stimuli is a possible pathophysiologic mechanism in functional dyspepsia (FD). Increasing evidence suggests involvement of both lateral and medial pain systems in normal visceral sensitivity and aberrant brain activation patterns in visceral hypersensitivity. We hypothesized that there is involvement of aberrant brain activation in FD with hypersensitivity to gastric distention. Our aim was to investigate regional cerebral blood flow during painful proximal gastric distention in hypersensitive FD. METHODS: Brain (15)O-water positron emission tomography was performed in 13 FD patients with symptoms of gastric hypersensitivity during 3 conditions: no distention, sham distention, and isobaric distention to unpleasant or painful sensation. Pain, discomfort, nausea, and bloating during maximal distention were rated on visual analogue scales. Data were analyzed using statistical parametric mapping. RESULTS: The threshold for painful distention was 6.6 +/- 3.8 mm Hg greater than the minimal distending pressure. At the corrected P level of less than .05, subtraction analysis (painful distention - no distention) showed activations in bilateral gyrus precentralis, bilateral gyrus frontalis inferior, bilateral gyrus frontalis medialis, bilateral gyrus temporalis superior, bilateral cerebellar hemisphere, and left gyrus temporalis inferior. Sham distention minus no distention showed no activations. CONCLUSIONS: Similar to healthy volunteers, proximal stomach distention in FD activates components of the lateral pain system and bilateral frontal inferior gyri, putatively involved in regulation of hunger and satiety. In hypersensitive FD, these activations occur at significantly lower distention pressures. In contrast to findings in normosensitivity, none of the components of the medial pain system were significantly activated.     

Gout, have we met before? No, not like this…

Extra-articular symptoms could be the first manifestation of gouty arthritis (GA); polyarticular GA can mimic an infectious arthritis; infection can complicate GA. A 66-year-old male with a history of gout presented with high fever and excruciating bilateral calf pain for 1 day. Examination revealed chronic knee effusions; range of motion in both knees was limited by calf pain. Joint aspiration showed negatively birefringent intracellular crystals and normal gram stain. While receiving empiric antibiotics fever continued and he developed bilateral knee, right ankle, and shoulder pain. After demonstration of urate crystals and exclusion of infection, antibiotics were discontinued and steroids initiated. Fever, calf pain, and polyarthritis quickly resolved. Polyarticular gouty attack is an uncommon presentation of gout, and can mimic several other conditions. An exceptional presentation of this entity is excruciating calf pain, probably caused by tenosynovitis or referred pain preceding an acute polyarticular gouty attack.     

Adult presentation of Stickler syndrome type III

Few clinical cases have been published on Stickler syndrome type III, and all describe the pediatric presentation. We describe an adult presentation of the syndrome in a 67-year-old woman and provide a report on the clinical and radiographic features supporting diagnosis. A chart review and updated investigations were performed to elucidate the presenting history and disease progression in the patient. Clinically, the patient was 42 years old when she presented with a 22-year history of bilateral knee pain and atypical osteoarthritis (OA) of an inflammatory but non-erosive character. The patient also reported increasing hearing loss for high-pitched frequencies. Radiography/X-rays at age 42 years showed severe, non-erosive tri-compartmental OA changes. Atypical destructive changes were identified in the small joints. The son of the patient developed left knee pain and bilateral knee swelling since the age of 9 years. He was diagnosed with early-onset OA and also underwent bilateral knee arthroplasties in his third decade, similar to his mother. The grandson presented at age 4 years, with recurrent, acute episodes of pain and swelling in his ankles and knees and was diagnosed with pediatric OA. A mutation of the COL11A2 gene was confirmed in the patient at aged 63 years. This is the first clinical case report on the adult presentation of Stickler syndrome type III. In particular, early-onset OA or an unrecognized skeletal dysplasia can be considered as rationale for genetic testing, screening, and surveillance of both past and present family members of an affected patient.     

Bilateral breast myxedema caused by Graves’ disease and responsive to multipoint subcutaneous injection of long-acting glucocorticoid: Case report

Rationale:With the absence of ophthalmopathy, thyroid dermopathy especially lesions at atypical locations is a very rare presentation. We herein report an original case of bilateral breast myxedema caused by Grave''s disease.Patient concerns:A 21-year-old unmarried woman presented with a 4-month history of Grave''s disease and a 1-month history of progressive bilateral breast enlargement. She had symmetrical bilateral breast enlargement with redness and nonpitting thickening of the skin, diffusely enlarged thyroid glands, and no exophthalmos.Diagnosis:Ultrasonography, magnetic resonance imaging scan, and skin biopsy confirmed the diagnosis of bilateral breast myxedema.Interventions:The patient was treated with multipoint subcutaneous injections of triamcinolone acetonide in each breast every month.Outcomes:The bilateral breast returned approximately to its normal size after therapy for 6 months.Conclusions:Our case illustrates that multipoint subcutaneous injection of glucocorticoids is beneficial for bilateral breast myxedema.     

Gout, have we met before? No, not like this..

LEARNING OBJECTIVES: Extra-articular symptoms could be the first manifestation of gouty arthritis (GA); polyarticular GA can mimic an infectious arthritis; infection can complicate GA. CASE: A 66-year-old male with a history of gout presented with high fever and excruciating bilateral calf pain for 1 day. Examination revealed chronic knee effusions; range of motion in both knees was limited by calf pain. Joint aspiration showed negatively birefringent intracellular crystals and normal gram stain. HOSPITAL COURSE: While receiving empiric antibiotics fever continued and he developed bilateral knee, right ankle, and shoulder pain. After demonstration of urate crystals and exclusion of infection, antibiotics were discontinued and steroids initiated. Fever, calf pain, and polyarthritis quickly resolved. DISCUSSION: Polyarticular gouty attack is an uncommon presentation of gout, and can mimic several other conditions. An exceptional presentation of this entity is excruciating calf pain, probably caused by tenosynovitis or referred pain preceding an acute polyarticular gouty attack.     

Hip Pain and Heart Failure: The Missing Link

A man presented with hypothyroidism, dilated cardiomyopathy, a pericardial effusion, liver failure, and polycythaemia. He had a history of bilateral hip replacements and new-onset hip pain. The patient progressed to develop shock. Given his acutely profound illness and constellation of symptoms, as well as the history of hip replacement, a diagnosis of cobalt toxicity was made.     

Atraumatic aseptic necrosis of the talus with varus deformity of the knee in an elderly patient

A 78-year-old woman was hospitalized with right ankle pain. Chronic pain had been present for 5 years without any history of trauma. Radiographs revealed osteosclerosis and depression at the talus. The patient also had osteoarthritis in the bilateral knees with varus deformity. We performed total knee arthroplasty and obtained good leg alignment. The ankle was treated by bracing and observed. Thereafter, ankle pain gradually decreased. Five years after surgery, the patient has no pain in her ankle and can walk about 1000 m without bracing. Radiograph revealed that the difference in talar articular surface was smoothly remodelled.     

Pseudomonas aeruginosa costovertebral arthritis in association with spontaneous cervical spondylodiscitis and epidural abscesses in the elderly

Cervical spondylodiscitis is an uncommon skeletal infection and its association with Pseudomonas aeruginosa has only been described in isolated case reports. Causes of cervical spondylodiscitis in these patients have been identified, except for the present case. A man aged 78 years with no significant previous medical history presented with an 8-week history of cervical pain and bilateral C5-7 radiculopathy. Magnetic resonance imaging revealed epidural abscesses and the destruction of C7 and T1 vertebrae, the interposing disc, and bilateral costovertebral joints. P aeruginosa was grown from open biopsy tissues and intravenous antibiotics were then administered to treat the infection. At the end of the 12-month follow-up period, all presenting symptoms had resolved and inflammatory markers (erythrocyte sedimentation rate and C-reactive protein) were within the normal ranges. Despite the infrequent incidence of cervical spondylodiscitis, it should be considered in elderly patients in whom risk factors are not found but unrelenting neck or back pain is reported.     

Adrenal tuberculosis: case report

Isolated adrenal tuberculosis is rare, and represents between 1-2% of the etiologies of adrenal masses called incidentalomas. A 32-year-old woman, without notable medical history, was hospitalized for pain in the left hypochondrium, lasting for two months in a context of apyrexia and weight loss amounted to 5 kg. Clinical examination was normal, but abdominopelvic CT objectified bilateral adrenal hypertrophy predominantly left with bilateral linear calcifications. The chest radiograph was normal, adrenal hormones were normal. The research of BK in sputum and urine were negative on direct examination and culture. The tuberculin was 12 mm and HIV status was negative. A left adrenal biopsy was done and histopathological study of tuberculous lesions was found confirming caseofolliculaire adrenal tuberculosis. The patient has been treated with antibacillaire with favorable evolution. In light of this observation, the authors make the point on this rare disease.     

Spontaneous bacterial empyema in a noncirrhotic patient: an unusual scenario

Spontaneous bacterial empyema (SBEM) is infection of a preexisting pleural effusion without evidence of pneumonia. It has been reported mostly in patients with hepatic hydrothorax. Only 1 case of SBEM in a noncirrhotic patient has been reported. We present an unusual case of bilateral SBEM from Streptococcus pneumoniae bacteremia in a noncirrhotic patient. A 52-year-old man presented with bilateral pleuritic chest pain and dyspnea for 2 days. His medical history included congestive heart failure, hemodialysis-dependent renal failure and known bilateral pleural effusions. No ascites or hepatosplenomegaly was noticed. Bilateral pleural effusions were again present on physical examination and confirmed by a chest computed tomography scan. Cardiac medical treatment and hemodialysis failed to improve his condition. Bilateral thoracentesis revealed purulent pleural fluid that was culture-positive for Streptococcus pneumonia as were blood cultures. There was no clinical or radiographic evidence of pneumonia. The detailed clinical course, treatment and highlighted points are described.     

IgG4-related autoimmune pancreatitis overlapping with Mikulicz’s disease and lymphadenitis:A case report

Autoimmune pancreatitis(AIP)is a form of chronic pancreatitis that is categorized as type 1 or type 2according to the clinical profile.Type 1 AIP,which predominantly presents in a few Asian countries,is a hyper-IgG4-related disease.We report a case of IgG4-related AIP overlapping with Mikulicz’s disease and lymphadenitis,which is rare and seldom reported in literature.A 63-year male from Northeast China was admitted for abdominal distension lasting for one year.He presented symmetric swelling of the parotid and submandibular glands with slight dysfunction of salivary secretion for 6 mo.He had a 2-year history of bilateral submandibular lymphadenopathy without pain.He underwent surgical excision of the right submandibular lymph node one year prior to admission.He denied any history of alcohol,tobacco,or illicit drug use.Serological examination revealed high fasting blood sugar level(8.8 mmol/L)and high level of IgG4(15.2 g/L).Anti-SSA or anti-SSB were negative.Computed tomography of the abdomen showed a diffusely enlarged pancreas with loss of lobulation.Immunohistochemical stain for IgG4 demonstrated diffuse infiltration of IgG4-positive plasma cells in labial salivary gland and lymph node biopsy specimens.The patient received a dose of 30 mg/d of prednisone for three weeks.At this three-week follow-up,the patient reported no discomfort and his swollen salivary glands,neck lymph node and pancreas had returned to normal size.The patient received a maintenance dose of 10mg/d of prednisone for 6 mo,after which his illness had not recurred.     

Carbamazepine and the lung

A 69 yr old man was admitted with a 10 day history of fever, arthromyalgia, dyspnoea, dry cough and pleuritic pain. Temperature was 38 degrees C; tachypnoea 36 rpm. Extensive crackles were audible over both upper lung fields. Chest X-ray showed bilateral alveolar infiltrates. Forced vital capacity was 49% of predicted, and carbon monoxide transfer coefficient was 32% of predicted value. The patient had been taking carbamazepine for one month because of a trigeminal neuralgia. After withdrawal of the drug he gradually recovered.     

The significance of Babinski signs in children with head trauma

A prospective study was undertaken to investigate the significance of Babinski signs in children with head trauma. Thirty-eight children between the ages of 1 and 15 were studied. Twenty-four of the children were admitted to the hospital for observation; 14 were sent home, with 24-hour follow-up in the pediatrics clinic. Of the 24 children admitted, six were believed to have altered CNS status or other focal findings on examination in the emergency department, and 18 were judged by two independent observers to have Babinski signs, either unilateral or bilateral, as their only neurological finding. Twelve hours after admission, all 18 children who had been admitted on the basis of Babinski signs alone had downgoing toes and they continued to have Babinski signs. The isolated presence of Babinski signs in a child with a history of head injury is not indicative of poor neurological outcome. An otherwise asymptomatic child who presents with a history of head injury and a solitary finding of Babinski sign(s) may be observed safely at home rather than being admitted to the hospital for observation.     

A challenging case of rheumatoid arthritis in an acromegalic patient

A 63-year-old man with complaints of joint pain and ankle swelling was evaluated. The arthralgias he described were mainly in the knees, elbows, and shoulders. Accompanying swelling and erythema in his left ankle and left second metacarpophalangeal (MCP) joint had recently ensued. His past history revealed acromegaly, somatotropinectomy, and radiotherapy. His neck, bilateral wrist, elbow, and shoulder joints were involved; there was pain and limited range of motion. The MCP joints, being worse than the interphalangeal joints, were likewise involved. His left ankle and MCP joints additionally were swollen and erythematous. Laboratory and radiological evaluations were carried out. Radiological and clinical findings confirmed a diagnosis of rheumatoid arthritis and concurrent acromegalic arthropathy. The patient was treated accordingly. Interestingly, he later developed colon cancer.     

耐甲氧西林金黄色葡萄球菌肺炎并发败血症及多处动脉瘤和动脉血栓一例报告并文献复习

目的提高对耐甲氧西林金黄色葡萄球菌（MRSA）肺炎并发败血症及动脉瘤和动脉血栓的认识。方法结合1例MRSA肺炎并发败血症及多处动脉瘤和动脉血栓患者的临床资料进行文献复习，对MRSA败血症的并发症、动脉瘤及动脉血栓的病理生理以及三者之间的内在联系进行分析。结果患者，男，66岁，因“发热、咳嗽、咯痰1个月余”于2006年8月16日入院。入院前发热1个月，入院时体温达40℃，入院后抗感染治疗10d左右体温降至正常，2次血培养及痰培养（痰液经镜检，均为标准痰）均为MRSA，诊断为院外感染的MRSA肺炎、败血症。静脉滴注万古霉素抗感染治疗后体温降至正常，但随后出现发热、疼痛、搏动性包块三联征，经B型超声及双下肢强化CT确诊为双侧髂外动脉、双侧臀上动脉多发性动脉瘤并附壁血栓形成、双侧股动脉及双侧股深动脉血栓形成等并发症。结论MRSA肺炎、败血症合并多发动脉瘤及动脉血栓非常少见，其原因可能是在动脉炎的基础上，内皮细胞损伤，引起了一系列凝血反应；血管肌层受损可导致或加重动脉扩张，形成动脉瘤。     

Spontaneous bilateral rupture of the Achilles tendon in a patient with chronic obstructive pulmonary disease

A 69-year-old man with chronic obstructive pulmonary disease (COPD) presented with an exacerbation of cough and breathlessness, as well as a 5 day history of sudden-onset bilateral calf tenderness. He had been commenced on inhaled steroids 41/2 years earlier and then received maintenance oral prednisone. Upon examination, there was a haematoma inferior to the medial malleolus with no Achilles tenderness on the left side. On the right side, there was focal tenderness over the mid-portion of the Achilles tendon with pain accentuated upon dorsi flexion. A venous duplex study confirmed superficial venous thrombosis involving the left gastrocnemius vein extending proximally to the popliteal vein junction. The major axial deep veins of the left lower leg were patent. Findings on the right side were normal. A subsequent diagnostic ultrasound demonstrated unequivocal bilateral Achilles tendon ruptures. The patient subsequently underwent corrective surgery. There have been several reports of bilateral Achilles tendon rupture associated with long-term corticosteroid use. It is likely that this entity is underdiagnosed because of a lack of awareness of this association by physicians. Recognition and surgical intervention are likely to reduce morbidity and improve outcome.