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 pa ents with primary hyperparathyroidism. It is also was reported in pa ents with sec ondary hyperparathyroidism due to end stage renal disease or Vitamin D dependent rickets .We present a case of celiac disease and osteomalacia which leads to secondary hyperparathyroidism and myelofibrosis. Keywords: Celiac disease, Hyperparathyroidism, Myelofibrosis
Introduction glands. Parathyroid carcinoma has been reported in Celiac sprue is an autoimmune disease caused pa ents with celiac disease. 9,10 In pa ents with celiac by gluten containing diet in gene cally predisposed disease with hypocalcemia and Vitamin D deficiency, persons. Pathogenesis is presence of an bodies to presence of hypophosphatemia associated with ssue transglutaminase, endomyosium, re culin, low to normal Calcium and elevated PTH level is and gliadin.1 It leads to malabsorp on of nutrients sugges ve of secondary hyperparathyroidism. 7 in small intes ne such as iron, folate, and calcium. 2 Literature review demonstrates that Presenta ons of pa ents 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 manifesta ons. pancytopenia whose bone marrow biopsy revealed Associated condi ons include derma s myelofibrosis. She underwent total excision of herpe formis, selec ve IgA deficiency, and other parathyroid adenoma .A er surgery, complete blood
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, thyroid disease, and liver slowly recovered without another interven on. 12 disease.1 In females with celiac disease, chronic Nomura et al reported a woman undergoing malabsorp on and nutri onal deficiency can alter hemodialysis with secondary hyperparathyroidism the func on of hypothalamic pituitary axis and lead who recovered from myelofibrosis a er a total to primary amenorrhea and failure of development parathyroidectomy. 4 of secondary sexual characteris cs. 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 pa ents with end stage myelofibrosis. renal disease, 4,5 vitamin D dependent rickets, 6 and rarely in celiac disease. 7 PTH ac vates osteoclasts, 8 Case Report increases renal clearance of phosphorus, bone An 18 year old girl was admi ed to hematology resorp on, serum levels of 1,25 dihydroxy vitamin department with pancytopenia and splenomegaly. D and intes nal absorp on of calcium, and She had posi ve history of chronic diarrhea since decreases renal clearance of calcium. 7 In pa ents childhood, and primary amenorrhea. Physical with celiac disease, hypocalcemia may persist examina on revealed short statue, clubbing, despite secondary hyperparathyroidism and lead splenpmegaly, and failure of development of to s mula on and enlargement of the parathyroid secondary sexual characteris cs. 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 pa ents 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, alkaline phosphatase of 654 U/L (high), have been reported. 12,13 In pa ents 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, par al thromboplas n me of 58 seconds, serum albumin of 4.3g/dl, erythrocyte References sedimenta on rate of 16 mm/h, ferri n of 20 ng/ 1 Kumar V, Rajadhyaksha M, Wortsman J. Celiac ml, normal iron and total iron binding capacity, disease associated autoimmune endocrinopathies. lactate dehydrogenase of 808 U/L (Nl<480), and Clin Diagn Lab Immunol. 2001; 8: 678 85. nega ve direct and indirect Coombs. ANA was 2 Elsurer R, Tatar G, Simsek H, Balaban YH, 0.4 IU/ml, calcium 7 mg/dl (normal: 8.5 10.3), Ay¬dinli M, Sokmensuer C. Celiac disease in phosphorus 1.5 mg/dl (normal: 1.7 4.5), potassium the Turkish popula on. Diges ve Diseases and 2.9 mEq/L, parathyroid hormone 226 pg/ml (high), Sciences. 2005; 50: 136 42. crea nine 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 func on tests were all normal. Serum folate level amenorrhea:a case report. J Reprod Med. 2007; was 2.03 ng/ml (normal: 3.1 17.5), vitamin B12 52: 453 5. level 176 pg/ml (normal: 191 663), prolac n 17 ng/ 4 Nomura S, Ogawa Y, Osawa G, Katagiri M, ml (normal), an endomyosial an body (Ig A) 170 Harada T, Nagahana H. Myelofibrosis secondary to U/ml (high). Abdominal ultrasonography revealed renal osteodystrophy. Nephron.1996; 72: 683 7. 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: 755 64. and hypochromia. Bone marrow aspira on showed 6 Stephan JL, Galambrun C, Dutour A, Freycon F. dry tap. A er many a empts, bone marrow was Myelofibrosis: an unusual presenta on of vitamin aspirated which showed severe hypocellolarity D deficient rickets. Eur J Pediatr. 1999; 158: 828 9. with increased number of osteoclasts, giant 7 Demay MB, Rosenthal DI, Deshpande V. Case cells, and osteoblasts. Bone marrow biopsy and records of the Massachuse s General Hospital. re culin staining revealed myelofibrosis. Upper Case 16 2008. A 46 Year Old Woman with Bone gastrointes nal endoscopy revealed fissures in the Pain. N Engl J Med. 2008; 358: 2266 74.
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 fla ening. Diagnosis of Yamaguchi K, Kinosaki M, Mochizuki S, et al. celiac disease and secondary hyperparathyroidism Osteoclast dif¬feren a on factor is a ligand for with myelofibrosis and central hypogonadism due osteoprotegerin/osteoclastogenesis inhibitory to systemic illness was made. We started gluten factor and is iden cal to TRANCE/RANKL. Proc Natl free diet and calcium, vitamins including vitamin D, Acad Sci USA. 1998; 95: 3597 3602. B12, C, E and folic acid. 9 Boyle NH, Ogg CS, Hartley RB, Owen WJ. Par¬athyroid carcinoma secondary to prolonged Discussion hyper¬plasia in chronic renal failure and in coeliac Pa ents with celiac disease and other disease. Eur J Surg Oncol. 1999; 25: 100 3. malabsorp on syndromes with vitamin D 10 Broadus AE, Braaten KM. Case records deficiency and hypocalcemia are suscep ble to of the Massachuse s General Hospital. Weekly secondary hyperparathyroidism which may lead to clinico¬pathological exercises. Case 7 2002. myelofibrosis. Bone marrow aspira on and biopsy A 47 year old woman with late recurrent are recommended in pa ents with celiac disease hyperparathyroidism. N Engl J Med. 2002; 346: who have pancytopenia, organomegaly, and elevated 694 700. PTH level. Searching for hyperparathyroidism should 11 Lim 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: 464 8. 12 Kumbasar B, Taylan I, Kazancioglu R, Agan M, Yenigun M, Sar F. Myelofibrosis secondary to hyperparathyroidism. Exp Clin Endocrinol Diabetes. 2004; 112: 127 30. 13 Bhadada SK, Bhansali A, Ahluwalia J, Chanukya GV, Behera A, Du a P. Anaemia and mar¬row fibrosis in pa ents with primary hyperparathy¬roidism before and a er cura ve parathyroidecto¬my. Clin Endocrinol (Oxf). 2000; 70: 527 32. Downloaded from ijbc.ir at 20:11 +0330 on Wednesday September 29th 2021
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