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Primary hypertrophic osteoarthropathy represents only about 3-5% of all cases of hypertrophic osteoarthropathy and is therefore a rare disease (1, 6). Characteristic X-ray findings are demonstrated for a specific case report.
Pachydermoperiostosis or primary hypertrophic osteoarthropathy is a rare disease characterized by cutaneous and osteoarthicular involvement. We describe two patients with finger clubbing, watch crystal nails, bones thickenings, arthritis and different grades of skin affection, without other clinical manifestations. Both did not know of having relatives with the same alterations. Radiological studies of the affected areas showed periostosis. Because of normal laboratory results and chest radiography plus the absence of other underlying causes, diagnosis of primary hypertrophic osteoarthropathy was made.
Hypertrophic osteoarthropathy is characterized by digital clubbing and periosteal reaction of long bones. Most cases are associated with malignancy or other conditions such as congenital heart disease, liver cirrhosis, pulmonary fibrosis, biliary atresia, and gastrointestinal polyps. We report a 19-year-old man presenting with arthritis, broadening of the fingers and clubbing of the fingers and toes for the previous 3 years. The ankles and knees were swollen. X-rays showed periosteal apposition. The search for a secondary cause remained negative. In cases of arthralgia/arthritis together with clubbed fingers, consideration must be given to hypertrophic osteoarthropathy. The primary or idiopathic form is rare and has a good prognosis.
A case of the very rare primary-hypertrophic-osteoarthropathy (Touraine-Solente-Golé-Syndrome) is reported. A middle-aged women was suffering from chronic joint- and limb-pain for more than twenty years. All diagnostical experiences only showed a cortical thickening mainly of the tubular bones and a craniosclerosis but no link for the possible origin. As typical for the disease, periosteal reactions could be discovered in X-ray examinations too. Thickening of the facial skin as well as drumstick-configurations of the fingers could be seen. A pulmonary affection leading to a secondary pulmonary osteoarthropathy (Pierre-Marie-Bamberger's Syndrome) could be excluded. The syndrome presented here is only to be found in 3 to 5% of all cases with hypertrophic osteoarthropathy. It is a rare differential-diagnosis of chronic bone and joint pain.
We describe seven patients with primary HOA and review 125 cases reported in the English, French, and German literature. The salient clinical features of primary HOA are: a bimodal distribution of disease onset with one peak during the first year of life and the other at age 15, a male predominance (nine to one), uncommon benign joint effusion, and a variety of skin abnormalities resulting from cutaneous hypertrophy or glandular dysfunction. We concluded that HOA is not a synovial disease. It is suggested that synovial effusions, when present, are perhaps a sympathetic reaction to the neighboring periostitis. Proposed diagnostic criteria for HOA, including digital clubbing and radiographic periostitis, appear 86% sensitive. The clinical features, age of onset, and sex distribution suggest that a genetically controlled growth promoting factor, different from growth hormone, plays a role in the pathogenesis of this syndrome.
This paper reports 2 cases of primary hypertrophic osteoarthopathy (PHO) which evolved into secondary hypertrophic osteoarthopathy (SHO) under the influence of bronchogenic carcinoma. The patients had a clinical picture of primary hypertrophic osteoarthropathy but without any signs of disease activity until in the last several months when a malignant bronchopulmonary condition developed. This activated all the symptoms: joint swelling; enlarged fingers and distal forearms and legs; moist palms and soles; unpleasant odour of perspiration; and deeper folds of the forehead and nasolabial furrow. A bone scan showed increased accumulation of the radioisotope in specific regions of the skeleton. To our knowledge, no similar cases have been described in the literature.
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Primary hypertrophic osteoarthropathy is a rare hereditary disease without evidence of underlying diseases. We report a very unusual case of primary HOA accompanied by Crohn's disease with the primary HOA mimicking secondary HOA, which is a rare manifestation of Crohn's disease. We also review the literature to find the correlation, if any, between the two.
A 37-year-old-patient was admitted to the medical polyclinic with a history of lower leg pain that had lasted for months. Pronounced clibbed fingers and toes were noted on clinical examination but other wise the patient's general condition was unremarkble. None of the routine tests performed evidenced pathological findings. Radiological examination revealed a lamellar periosteal reaction along both sides of the tibia and fibula consistent with hypertrophic osteoarthropathy. A computed tomogram taken of the thorax to exclude a tumor was inconspicuous. Treatment with nonsteroid antirheumatics resulted in complete eradication of the symptoms. In case of uncertain bone pain together with clubbed fingers, consideration must be given to hypertrophic osteoarthropathy. This condition is usually a secondary manifestation, especially in cases of cardiac or pulmonary disease. The primary or idiopathic forms is rare and has a good prognosis.
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In this article a family with five members suffering from primary hypertrophic osteoarthropathy (PHO) is reported. Our cases are characterized by swollen and painful joints and clubbing of distal phalanges of hands and feet. The diagnosis made upon on radiological and clinical findings.
Cutaneous fibrinolytic activity was investigated in 13 patients affected with Primary Hypertrophic Osteoarthropathy in the advanced phase. Biopsies were taken from periungueal skin of the ippocratic finger and from the dorsum of the first phalanx: cutaneous fibrinolytic activity was investigated with Todd's autohistographic method as modified by Lotti et al., showing increased activity in periungueal skin (10/13) and normal in the apparently uninvolved skin (9/13). We suggest that the increase in plasminogen activator activity, in the advanced phase of the disease, may be related to the increased growth of the connective tissue that is characteristic of the disease.
BACKGROUND: Our aim was to establish clinical and imaging characteristics of primary hypertrophic osteoarthropathy (PHO). METHODS: A group of 76 patients (71 men and 5 women) with primary hypertrophic osteoarthropathy was examined. Extensive history and status of the locomotor system were determined in all patients. Radiologic and scintigraphic examinations of the bones, thermography, capillaroscopy, gastroscopy, and histologic analysis were also performed. RESULTS: Family history was positive in all 55 patients (100%) summoned for a check-up. Finger clubbing of the hands and feet as well as positive Schamroth test were found in all patients, while hyperhydrosis was found in 65 (85.5%), seborrhea in 39 (51%), and involvement of the joints in 52 (68.5%) patients. Histologic analysis in 18 (23.7%) patients predominantly showed periarticular edema and moderate cellular activity. Periostosis of the long bones was also found in all patients, while scintigraphy, performed in 44 patients, was positive in 18 (33.5%) patients in the active phase of the disease. Thermography, performed in 10 patients, ranged from hypothermia to thermal amputation of feet and hands. Capillaroscopy of hands and feet in 47 (61.8%) patients showed changes in small blood vessels. Gastroscopy indicated abnormalities (ulcer and hypertrophic gastritis) in 21 patients (27.6%). CONCLUSIONS: PHO has special clinical and radiographic manifestations (finger clubbing and periostosis of the long bones) that are associated with some minor manifestations. Development of the disease is gradual (to 20 years) and requires a special diagnostic assessment.
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