See discussions, stats, and author profiles for this publication at: https://www.researchgate.net/publication/13912216 Hypophosphatemic Rickets with Sacroiliitis-like Presentation in an Adolescent Article in Scandinavian Journal of Rheumatology · February 1997 DOI: 10.3109/03009749709105326 · Source: PubMed CITATIONS READS 3 109 6 authors , including: Reyhan Celiker Alp Cetin Ac ı badem Mehmet Ali Ayd ı nlar Üniversitesi Hacettepe University 66 PUBLICATIONS 1,125 CITATIONS 49 PUBLICATIONS 668 CITATIONS SEE PROFILE SEE PROFILE Ayfer Alikasifoglu Hacettepe University 95 PUBLICATIONS 944 CITATIONS SEE PROFILE Some of the authors of this publication are also working on these related projects: Obesity View project All content following this page was uploaded by Reyhan Celiker on 20 January 2015. The user has requested enhancement of the downloaded file.
J Rheumatol1997;26:332-5 zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA Scund zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA in an Adolescent zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA CASE REPORT zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA Hypophosphatemic Rickets with Sacroiliitis-like Presentation Medicine, Ankara, Turkey zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA 0. Onur', R. Celiker', A. Cetin', A. Alikagifoglu', 0. Ugur3, and 0. Baggoze' Hacettepe University School of Medicine, 'Department of Physical Medicine and Rehabilitation, 'Department of Pediatrics, 3Department of Nuclear Rickets can manifest with a wide variety of rheumatic symptoms. In this paper, a fifteen year old female patient with hypophosphatemic rickets presenting with symptoms suggesting sacroiliitis at disease onset is reported. The sacroiliac joint involvement in this case was attributed to the subchondral bone fractures due to the secondary hyperparathyroidism. Her symptoms resolved completely after treatment with calcitriol and phosphate solution for hypophosphatemic rickets Key words: hypophosphatemic rickets, sacroiliac joint Scand J Rheumatol Downloaded from informahealthcare.com by HINARI on 01/20/15 proximal muscles of the lower extremities (4/5 in Rickets has largely been eliminated as a medical manual muscle testing according to the Medical concern even in most of the underdeveloped countries because of Vitamin D supplementation and exposure Research Council) and she had a waddling gait. to the sun. As a result, the cases that are seen are Values for hemoglobin, white blood cell count, eryth- more likely to have a genetic basis. Familial hypopho- rocyte sedimentation rate, all measured with routine sphatemic rickets, previously known as Vitamin D techniques, were normal ( 134 gr/L, 4.80 x 103/mm3 resistant rickets, appears to be the most common and 16 mm/h respectively). Antistreptolysin-0 Anti- cause of inherited rickets ( 1 ). body (ASO), C-reactive protein and rheumatoid For personal use only. Rickets can manifest with a wide variety of symp- factor measured with standard serologic tests were toms, principally with bone pain and joint involve- negative. Serum alkaline phosphatase level was elev- ment, that may be mistaken for a rheumatic disease ated to 2235U/L (normal range: 70-230U/L in 14-15 year old children). She was normocalcemic but (2). We report an unusual case of hypophosphatemic hypophosphatemic with a serum calcium level of rickets in an adolescent female patient. She had 2.32mmol/L and a serum phosphorous level of symptoms suggesting sacroiliac joint involvement as 0.45mmol/L (normal range for serum calcium and the first manifestation of the disease. phosphorous levels are 2.12-2.62 mmol/L and 0.96-1.45 mmol/L respectively). The TmP04/GF was 0.36 mmol/L which is far below the normal Case Report range. Renal function studies including BUN, crea- A fifteen year old girl was referred to Hacettepe tinine, and renal aminoacid excretion were normal. University Hospital, Department of Physical Parathyroid hormone level, measured by specific ami- Medicine and Rehabilitation, with a complaint of noterminal radioimmunoassay was 370 pg/mL pain in her hips and low back region. She had been (normal range is 10-65 pg/mL). Serum 25(OH)Vita- in pain for 6 months and the pain had worsened min D, level was 45 nmol/L which is in the normal gradually during the last 3 months. She also com- range of 25-100 nmol/L. plained of fatigue and weakness and with difficulty Complete skeletal X rays were obtained. X rays of in climbing stairs and standing up from seated the skull, hands and long bones revealed a normal position. bone density and appearance. However, radiological Physical examination revealed pain in the hip joints examination of the lumbosacral region demonstrated and low back region in the extremes of motion, pain ill-defined and in some regions, sclerotic sacroiliac joints. Sacroiliac X rays confirmed the sacroiliac joint on sacroiliac compression tests and positive Fabere Accepted zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA 332 zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA test on both sides. There was a mild weakness in involvement (Fig. 1 ). A total body radionucleide bone imaging with 15 mCi 99mTc-methylene bisphosphonate revealed Reyhan Celiker, Gerede Sokak 417, 06540, Agagi Ayranci, Ankara, diffuse radioactive uptake in all bones of the body Turkey including the mandible, pelvic bones, and the verteb- Received 12 June 1996 rae (Fig. 2). The proximal parts of both femur and 4 April 1997 1997 Scandinavian University Press on license from Scandinavian Rheumatology Research Foundation 8
3. zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA activity uptake in the zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA (posterior view) zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA Frg. zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA Fig. zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA Scintigraphic examination demonstrating increased radio- Scand J Rheumatol Downloaded from informahealthcare.com by HINARI on 01/20/15 left sacroiliac joint suggesting left sacroiliitis An ultrasonographic examination of parathyroid 1. X ray examination of lumbosacral region demonstrating glands was normal. ill-defined sacroiliac joints. 25(OH )Vitamin D, level. hypophosphatemia, and zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA The motor and sensory nerve conduction studies and needle electromyographic investigation of the the ribs showed an irregular radioactivity. The left lower extremities were normal. Although the primary complaint. physical findings, sacroiliac joint had a greater radioactive uptake than For personal use only. and radiological examination of the patient were the right side, with an index of 1.50 and 1.28 respect- ively. This is above the normal accepted range on the consistent with sacroiliitis, the biochemical para- left side (Fig. 3). As a result scintigrafic investigation meters and the scintigraphy suggested a metabolic bone disease as well. Normal serum calcium and suggested a metabolic bone disease and left sacroiliitis. $5-%4-% 19: zyxwvutsrqponmlkjihgfedcbaZYXWVUTSRQPONMLKJIHGFEDCBA 39 Fig. 2. Total body radionucleide imaging demonstrating dif-rusr radioactive uptake of all bonea including the mandible. vertebrae. costochondral junctions. and the pelvis suggesting a metabolic bone disease. 333
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