Department of Physical Medicine and Rehabilitation, Erciyes University Medical School, Kayseri, Turkey.
Hormones (Athens). 2013 Apr-Jun;12(2):309-11. doi: 10.14310/horm.2002.1416.
An 18-year-old man was admitted to the clinic complaining of deterioration in the function of his hands and feet. The clinical examination revealed that his movements were clumsy and that he had disproportionally short limbs. In addition, he also had facial abnormalities of frontal bossing, hypertelorism, maxillary hypoplasia, broad low nasal bridge, short upturned nose with anteverted nostrils and triangular mouth. All extremities appeared short with stubby fingers and toes and with broad hands and wrinkling of the dorsal skin. Chromosomal analysis showed a normal (46, XY) karyotype. X-ray studies revealed broad, short metacarpals and phalanges with cone-shaped epiphyses and brachycdactyly and a diagnosis of peripheral dysostosis was confirmed by the characteristic radiographic appearance of the hands. Serum calcium and alkaline phosphatase levels were high, parathormone (PTH) was low, but 25 (OH) Vitamin D, albumin, and 24 hour urine calcium levels were in the normal range. Based on these findings, a diagnosis of acrodysostosis associated with hypercalcemia was made. To the best of our knowledge, this represents the first description of this syndrome.
一名 18 岁男性因手脚功能恶化到诊所就诊。临床检查发现他的动作笨拙,四肢不成比例地短小。此外,他还具有额骨突出、眼距过宽、上颌骨发育不全、宽而低的鼻梁、短而朝天的鼻子和前鼻孔翻转、三角口等面部异常。所有四肢都显得短而粗短的手指和脚趾,手宽,背部皮肤起皱。染色体分析显示正常的(46,XY)核型。X 射线研究显示宽而短的掌骨和指骨,伴有锥形骨骺和短指畸形,手部特征性的放射影像表现证实了外周骨发育不良的诊断。血清钙和碱性磷酸酶水平升高,甲状旁腺激素(PTH)降低,但 25(OH)维生素 D、白蛋白和 24 小时尿钙水平在正常范围内。基于这些发现,诊断为伴高钙血症的肢端发育不良。据我们所知,这是该综合征的首次描述。