Hypertrophic osteoarthropathy is characterized by clubbing and periosteal new bone formation along the shaft of the long bones of the extremities. Although various intrathoracic malignancies have been associated with the development of HOA, it has been extremely rare for HOA to occur in a patient with a thymic carcinoma. Recently, we experienced a 63-year-old woman diagnosed as a thymic carcinoma with hypertrophic osteoarthropathy. She had both digital clubbing and cortical thickening in her lower extremities identified radiologically. We herein describe this case with a review of the literature.
Carcinoma/complications/*diagnosis ; Female ; Human ; Middle Aged ; Osteoarthropathy, Secondary Hypertrophic/*etiology ; Thymus Neoplasms/complications/*diagnosis

Hypertrophic osteoarthropathy(HOA) is a systemic disorder primary affecting the bones, joints, and soft tissues and characterized by several(or all) of the followings ; 1) Clubbing of digits, 2) Persistent new bone formation particulary involving long bones of the distal extremites, 3) Symmetric arthritis-like changes in the joints and periarticular tissue, most commonly the ankles, knees, wrist, and elbows, 4) Increased thickness of the subcutaneous soft tissues in the distal one-third of the arms and legs, and 5) Neurovascular changes of the hands and feet, including chronic erythema, paresthesis, and increase sweating. Most of cases of HOA are secondary to intrathoracic neoplasms, while the remaining few cases are secondary to other disease in the chest or elsewhere. We experienced a case of HOA in association with lung abscess in 26-yr-old male and reported with a review of literatures.
Ankle ; Arm ; Elbow ; Erythema ; Foot ; Hand ; Humans ; Joints ; Knee ; Leg ; Lung Abscess* ; Lung* ; Male ; Osteoarthropathy, Primary Hypertrophic ; Osteoarthropathy, Secondary Hypertrophic* ; Osteogenesis ; Sweat ; Sweating ; Thorax ; Wrist

Three cases of linear scleroderma with typical clinical and histopathological findings were presented. Case 1: A 10 year old female had a "coupe de sabre" type of linear scleroderma on the right side of the frontal scalp and face and hemiatrophy of the right side of the face. Case 2: A 10 year old female had a type of scleroderma en bande on the palm and dorsum of the left hand and clubbed finger of the left third finger. Case 3: A 21 year old male had a linear type of skin atrophy and sclerosis of the right lower exteremity and mild walking disturbance.
Atrophy ; Child ; Female ; Fingers ; Hand ; Humans ; Male ; Osteoarthropathy, Secondary Hypertrophic ; Scalp ; Scleroderma, Localized ; Sclerosis ; Skin ; Walking ; Young Adult

Three cases of linear scleroderma with typical clinical and histopathological findings were presented. Case 1: A 10 year old female had a "coupe de sabre" type of linear scleroderma on the right side of the frontal scalp and face and hemiatrophy of the right side of the face. Case 2: A 10 year old female had a type of scleroderma en bande on the palm and dorsum of the left hand and clubbed finger of the left third finger. Case 3: A 21 year old male had a linear type of skin atrophy and sclerosis of the right lower exteremity and mild walking disturbance.
Atrophy ; Child ; Female ; Fingers ; Hand ; Humans ; Male ; Osteoarthropathy, Secondary Hypertrophic ; Scalp ; Scleroderma, Localized ; Sclerosis ; Skin ; Walking ; Young Adult

Metastatic carcinoma on the hand occurs in 0.1% of all metastasis and the diagnosis is difficult since it frequently mimics other conditions such as infection, osteomyelitis, gout, and pseudoclubbing. A 70-year-old woman with lung carcinoma was consulted for the evaluation of a painful swelling patch on the tip of her right 2nd finger. She was initially treated with antibiotics due to the misdiagnosis of the condition as an infection. Her pain persisted for a month. The skin biopsy revealed a metastatic adenocarcinoma with papillary growth pattern. We report a rare and an interesting metastatic carcinoma manifesting as clubbed finger.
Adenocarcinoma ; Aged ; Anti-Bacterial Agents ; Biopsy ; Diagnostic Errors ; Female ; Fingers ; Gout ; Hand ; Humans ; Lung ; Lung Neoplasms ; Neoplasm Metastasis ; Osteoarthropathy, Secondary Hypertrophic ; Osteomyelitis ; Skin

Pulmonary arteriovenous fistulas(PAVFs) is a rare disorder that occurs in two to three children per 100,000 population. It is presented as absence of intervening capillary beds between the pulmonary artery and vein with resultant persistent right to left shunt. Other causes include trauma, liver cirrhosis, malignancy and schistosomiasis. It is mostly asymptomatic, but it may present with respiratory difficulty, cyanosis, clubbed fingers induced by right to left shunt or hemoptysis, polycythemia and epistaxis. Major complications, such as brain abscess, brain embolism, paradoxical embolism and subacute infective endocarditis can be devastating, so therapeutic intervention is recommended in all patients. However, removal of low-resistance fistulas can aggrevate pulmonary hypertension, so detection of increased pulmonary pressure is important. We report two patients:One a 42 year-old male with PAVFs treated with coil embolization, and a 42 year-old female who was treated with anticoagulants due to pulmonary hypertension.
Adult ; Anticoagulants ; Arteriovenous Fistula* ; Brain Abscess ; Capillaries ; Child ; Cyanosis ; Embolism, Paradoxical ; Embolization, Therapeutic ; Endocarditis ; Epistaxis ; Female ; Fistula ; Hemoptysis ; Humans ; Hypertension ; Hypertension, Pulmonary* ; Intracranial Embolism ; Liver Cirrhosis ; Male ; Osteoarthropathy, Secondary Hypertrophic ; Polycythemia ; Pulmonary Artery ; Schistosomiasis ; Veins

Hepatopulmonary syndrome (HPS) is a serious complication of end-stage liver disease, which is characterized by hypoxia, intrapulmonary vascular dilatation, and liver cirrhosis. Liver transplantation (LT) is the only curative treatment modality for patients with HPS. However, morbidity and mortality after LT, especially in cases of severe HPS, remain high. This case report describes a patient with typical findings of an extracardiac pulmonary arteriovenous shunt on contrast-enhanced transesophageal echocardiography (TEE), and clubbing fingers, who had complete correction of HPS by deceased donor LT. The patient was a 16-year-old female who was born with biliary atresia and underwent porto-enterostomy on the 55th day after birth. She had been suffered from progressive liver failure with dyspnea, clubbing fingers, and cyanosis. Preoperative arterial blood gas analysis revealed severe hypoxia (arterial O2 tension of 54.5 mmHg and O2 saturation of 84.2%). Contrast-enhanced TEE revealed an extracardiac right-to-left shunt, which suggested an intrapulmonary arteriovenous shunt. The patient recovered successfully after LT, not only with respect to physical parameters but also for pychosocial activity, including school performance, during the 30-month follow-up period.
Adolescent ; Anoxia ; Arteriovenous Fistula/etiology ; Biliary Atresia/*diagnosis/etiology ; Cyanosis/complications ; Dyspnea/complications ; Echocardiography, Transesophageal ; End Stage Liver Disease/complications/*surgery ; Female ; Hepatic Artery/abnormalities ; Hepatopulmonary Syndrome/*diagnosis/ultrasonography ; Humans ; *Liver Transplantation ; Osteoarthropathy, Secondary Hypertrophic/complications

Epithelioid hemangioendothelioma is a rare vascular tumor, which occurs in the lung, liver, bone, and soft tissue. Hypertrophic osteoarthropathy is a syndrome characterized by subperiosteal new bone formation, joint effusion and clubbing, and may be associated with cyanotic heart disease, chronic pulmonary disease, liver disease, and other miscellaneous diseases. The activation of endothelium and platelets has been suggested to be involved in the development of hypertrophic osteoarthropathy. We report a rare case of hypertrophic osteoarthropathy, which developed in association with hepatic epithelioid hemangioendothelioma with pulmonary metastasis. We also discuss the role of vascular endothelial growth factor in its pathogenesis.
Adult ; Biopsy ; Blood Platelets/metabolism ; Endothelium/metabolism ; Femur/radiography ; Hemangioendothelioma, Epithelioid/*complications/*pathology ; Human ; Lung/pathology ; Lung Neoplasms/*complications/pathology ; Male ; Osteoarthropathy, Secondary Hypertrophic/*complications/*pathology ; Vascular Endothelial Growth Factor A/metabolism ; Vascular Neoplasms/*diagnosis/pathology

Hypertrophic osteoarthropathy is characterized by clubbing of the digital tips and periosteal reaction of long bones. Most of the cases are associated with malignancy or other conditions such as congenital heart disease, liver cirrhosis, pulmonary fibrosis, biliary atresia, and gastrointestinal polyps. Hypertrophic osteoarthropathy associated with malignancy is rare in children. A few cases of hypertrophic osteoarthropathy in children with nasopharyngeal carcinoma have been reported, however, there has been no report of such case in Korea. We present a case of hypertrophic osteoarthropathy associated with nasopharyngeal carcinoma with lung metastasis in a 14-yr-old boy. In this case, hypertrophic osteoarthropathy regressed after intensive chemotherapy, but subsequently the patient died of progressive lung metastasis.
Adolescent ; Bone and Bones/radiography ; Carcinoma/*diagnosis/mortality ; Disease Progression ; Fatal Outcome ; Human ; Joint Diseases/pathology ; Lung Neoplasms/mortality/*secondary ; Male ; Nasopharyngeal Neoplasms/*diagnosis/mortality ; Neoplasm Metastasis ; Osteoarthropathy, Primary Hypertrophic/*diagnosis/mortality/radiography ; Prognosis