CASE REPORT

IJBC 2010;3: 141-143 Myelofibrosis due to Secondary Hyperparathyroidism in a Case of Celiac Disease Nasim Valizadeh1, Neda Valizadeh1, Shahin Nateghi1, Negar Aghamohammadi1

1. Urmia University of Medical Sciences,Urmia, Iran; Corresponding author: Nasim Valizadeh, Assistant Professor of Hematology/oncology ; Urmia University of Medical Sciences,Urmia, Iran; (Tel/Fax: 09125474755, Email:[email protected])

Abstract Myelofibrosis is reported in paents with primary hyperparathyroidism. It is also was reported in paents with sec ondary hyperparathyroidism due to endstage renal disease or dependent .We present a case of celiac disease and which leads to secondary hyperparathyroidism and myelofibrosis. Keywords: Celiac disease, Hyperparathyroidism, Myelofibrosis

Introduction glands. has been reported in Celiac sprue is an autoimmune disease caused paents with celiac disease. 9,10 In paents with celiac by glutencontaining diet in genecally predisposed disease with and Vitamin D deficiency, persons. Pathogenesis is presence of anbodies to presence of associated with ssue transglutaminase, endomyosium, reculin, low to normal and elevated PTH level is and gliadin.1 It leads to malabsorpon of nutrients suggesve of secondary hyperparathyroidism. 7 in small intesne such as iron, folate, and calcium. 2 Literature review demonstrates that Presentaons of paents with celiac disease are hyperparathyroidism is an important cause of different including chronic diarrhea, short statue, myelofibrosis. 11 Kumbasar et al reported a young iron deficiency anemia, osteomalacia, neurologic female with primary hyperparathyroidism and problems, and other unusual manifestaons. pancytopenia whose bone marrow biopsy revealed Associated condions include dermas myelofibrosis. She underwent total excision of herpeformis, selecve IgA deficiency, and other .Aer surgery, complete

Downloaded from ijbc.ir at 20:11 +0330 on Wednesday September 29th 2021 diseases which have autoimmune bases such as count (CBC) and other clinical abnormal findings type 1 diabetes mellitus, disease, and liver slowly recovered without another intervenon. 12 disease.1 In females with celiac disease, chronic Nomura et al reported a woman undergoing malabsorpon and nutrional deficiency can alter hemodialysis with secondary hyperparathyroidism the funcon of hypothalamicpituitary axis and lead who recovered from myelofibrosis aer a total to primary amenorrhea and failure of development . 4 of secondary sexual characteriscs. 3 Secondary Here, we present a case of celiac disease with hyperparathyroidism is a physiologic response to osteomalacia, secondary hyperparathyroidism, and hypocalcemia and is seen in paents with end stage myelofibrosis. renal disease, 4,5 vitamin Ddependent rickets, 6 and rarely in celiac disease. 7 PTH acvates osteoclasts, 8 Case Report increases renal clearance of phosphorus, bone An 18yearold girl was admied to hematology resorpon, serum levels of 1,25dihydroxy vitamin department with pancytopenia and splenomegaly. D and intesnal absorpon of calcium, and She had posive history of chronic diarrhea since decreases renal clearance of calcium. 7 In paents childhood, and primary amenorrhea. Physical with celiac disease, hypocalcemia may persist examinaon revealed short statue, clubbing, despite secondary hyperparathyroidism and lead splenpmegaly, and failure of development of to smulaon and enlargement of the parathyroid secondary sexual characteriscs. Laboratory

IRANIAN JOURNAL OF BLOOD AND CANCER Volume 2 Number 3 Spring 2010 141 Nasim Valizadeh findings included leukocyte count of 1400/mm 3 be recommended in paents with celiac disease (polymorphonuclear= 54%, lymphocyte= 35%), and myelofibrosis. Many cases of myelofibrosis due hemoglobin of 7.1 g/dl, platelet count 52,000/ to primary and secondary hyperparathyroidism mm 3, of 654 U/L (high), have been reported. 12,13 In paents with primary total billirubin of 1.35 mg/dl (normal<1.2), direct hyperparathyroidism, total excision of parathyroid billirubin of 0.45 mg/dl (normal<0.2), prothrombin adenoma improves myelofibrosis. 9,11,12 me of 15 seconds, paral thromboplasn me of 58 seconds, serum albumin of 4.3g/dl, erythrocyte References sedimentaon rate of 16 mm/h, ferrin of 20 ng/ 1Kumar V, Rajadhyaksha M, Wortsman J. Celiac ml, normal iron and total iron binding capacity, diseaseassociated autoimmune endocrinopathies. lactate dehydrogenase of 808 U/L (Nl<480), and Clin Diagn Lab Immunol. 2001; 8: 67885. negave direct and indirect Coombs. ANA was 2Elsurer R, Tatar G, Simsek H, Balaban YH, 0.4 IU/ml, calcium 7 mg/dl (normal: 8.510.3), Ay¬dinli M, Sokmensuer C. Celiac disease in phosphorus 1.5 mg/dl (normal: 1.74.5), potassium the Turkish populaon. Digesve Diseases and 2.9 mEq/L, 226 pg/ml (high), Sciences. 2005; 50: 13642. creanine 0.7 mg/dl, and blood urea nitrogen 3 Pradhan M, Manisha, Singh R, Dhingra normal. Urine analysis, stool exam, and thyroid S. Celi¬ac disease as a rare cause of primary funcon tests were all normal. Serum folate level amenorrhea:a case report. J Reprod Med. 2007; was 2.03 ng/ml (normal: 3.117.5), vitamin B12 52: 4535. level 176 pg/ml (normal: 191663), prolacn 17 ng/ 4 Nomura S, Ogawa Y, Osawa G, Katagiri M, ml (normal), anendomyosial anbody (Ig A) 170 Harada T, Nagahana H. Myelofibrosis secondary to U/ml (high). Abdominal ultrasonography revealed . Nephron.1996; 72: 6837. hepatomegaly and huge splenomegaly (span=210 5 Weinberg SG, Lubin A, Wiener SN, Deoras MP, mm). Peripheral blood smear showed anisocytosis, Ghose MK, Kopelman RC. Myelofibrosis and renal poikylocytosis, macrocyte, microcyte, tear drop, osteodystrophy. Am J Med. 1977; 63: 75564. and hypochromia. Bone marrow aspiraon showed 6Stephan JL, Galambrun C, Dutour A, Freycon F. dry tap. Aer many aempts, bone marrow was Myelofibrosis: an unusual presentaon of vitamin aspirated which showed severe hypocellolarity Ddeficient rickets. Eur J Pediatr. 1999; 158: 8289. with increased number of osteoclasts, giant 7 Demay MB, Rosenthal DI, Deshpande V. Case cells, and osteoblasts. Bone marrow biopsy and records of the Massachuses General Hospital. reculin staining revealed myelofibrosis. Upper Case 162008. A 46YearOld Woman with Bone gastrointesnal endoscopy revealed fissures in the Pain. N Engl J Med. 2008; 358: 226674.

Downloaded from ijbc.ir at 20:11 +0330 on Wednesday September 29th 2021 second part of duodenum. Biopsy revealed marked 8 Yasuda H, Shima N, Nakagawa N, villous atrophy and mucosal flaening. Diagnosis of Yamaguchi K, Kinosaki M, Mochizuki S, et al. celiac disease and secondary hyperparathyroidism Osteoclast dif¬ferenaon factor is a ligand for with myelofibrosis and central hypogonadism due osteoprotegerin/osteoclastogenesisinhibitory to systemic illness was made. We started gluten factor and is idencal to TRANCE/RANKL. Proc Natl free diet and calcium, vitamins including vitamin D, Acad Sci USA. 1998; 95: 35973602. B12, C, E and folic acid. 9Boyle NH, Ogg CS, Hartley RB, Owen WJ. Par¬athyroid carcinoma secondary to prolonged Discussion hyper¬plasia in chronic renal failure and in coeliac Paents with celiac disease and other disease. Eur J Surg Oncol. 1999; 25: 1003. malabsorpon syndromes with vitamin D 10Broadus AE, Braaten KM. Case records deficiency and hypocalcemia are suscepble to of the Massachuses General Hospital. Weekly secondary hyperparathyroidism which may lead to clinico¬pathological exercises. Case 72002. myelofibrosis. Bone marrow aspiraon and biopsy A 47yearold woman with late recurrent are recommended in paents with celiac disease hyperparathyroidism. N Engl J Med. 2002; 346: who have pancytopenia, organomegaly, and elevated 694700. PTH level. Searching for hyperparathyroidism should 11Lim DJ, Oh EJ, Park CW. Pancytopenia and

142 IRANIAN JOURNAL OF BLOOD AND CANCER Myelofibrosis due to Secondary Hyperparathyroidism in a Case of Celiac Disease secondary myelofibrosis could be induced by pri¬mary hyperparathyroidism. Int J Lab Hematol. 2007; 29: 4648. 12Kumbasar B, Taylan I, Kazancioglu R, Agan M, Yenigun M, Sar F. Myelofibrosis secondary to hyperparathyroidism. Exp Clin Endocrinol Diabetes. 2004; 112: 12730. 13Bhadada SK, Bhansali A, Ahluwalia J, Chanukya GV, Behera A, Dua P. Anaemia and mar¬row fibrosis in paents with primary hyperparathy¬roidism before and aer curave parathyroidecto¬my. Clin Endocrinol (Oxf). 2000; 70: 52732. Downloaded from ijbc.ir at 20:11 +0330 on Wednesday September 29th 2021

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