Precursor T-cell acute lymphoblastic leukemia presenting with bone marrow necrosis: a case report

Journal of Medical Case Reports

Beston F Nore²  Hisham A Al-Rawi³  Najmaddin SH Khoshnaw¹ 

[1] Department of Hematology, Hiwa Hematology-Oncology Center, Kurdistan Regional Government, Sulaimaniyah, Iraq;Department of Biochemistry, Faculty of Medical Sciences, School of Medicine, University of Sulaimani, Kurdistan Regional Government, Sulaimaniyah, Iraq;Department of Hematopathology, University of Sulaimani, Kurdistan Regional Government, Sulaimaniyah, Iraq

关键词: Bone marrow aspiration;    Acute lymphoblastic leukemia;    Bone marrow necrosis;   

Others  :  1195342
DOI  :  10.1186/1752-1947-6-349

 received in 2012-05-05, accepted in 2012-08-29,  发布年份 2012

【 摘 要 】

Introduction

Bone marrow necrosis is a clinicopathological condition diagnosed most often at postmortem examination, but it is also seen during the course of malignancy and is not always associated with a poor prognosis. The morphological features of bone marrow necrosis are disruption of the normal marrow architecture and necrosis of myeloid tissue and medullary stroma. Non-malignant conditions associated with bone marrow necrosis are sickle cell anemia, infections, drugs (sulfasalazine, interferon α, all-trans retinoic acid, granulocyte colony-stimulating factor and fludarabine), disseminated intravascular coagulation, antiphospholipid antibody syndrome and acute graft versus host diseases. The malignant causes are leukemia, lymphoma and metastatic carcinomas. Herein we report the case of a patient with precursor T-cell acute lymphoblastic leukemia and bone marrow necrosis at initial presentation.

Case presentation

A 10-year-old Kurdish boy was presented with generalized bone pain and fever of 1 month’s duration which was associated with sweating, easy fatigability, nose bleeding, breathlessness and severe weight loss. On examination, we observed pallor, tachypnea, tachycardia, low blood pressure, fever, petechial hemorrhage, ecchymoses, tortuous dilated veins over the chest and upper part of abdomen, multiple small cervical lymph node enlargements, mildly enlarged spleen, palpable liver and gross abdominal distention. Blood analysis revealed pancytopenia and elevated lactate dehydrogenase and erythrocyte sedimentation rate. Imaging results showed mediastinal widening on a planar chest X-ray and diffuse focal infiltration of the axial bone marrow on magnetic resonance imaging of the lumbosacral vertebrae. Bone marrow aspiration and biopsy examination showed extensive bone marrow necrosis. Immunophenotyping analysis of the bone marrow biopsy confirmed T-cell acute lymphoblastic leukemia, as CD3 and terminal deoxynucleotidyl transferase markers were positive and CD10, CD20 and CD79a markers were negative.

Conclusion

The aggressive initial clinical presentation of our patient with huge mediastinal widening, development of superior vein cava syndrome and extensive bone marrow necrosis as initial signs made the diagnosis of the case difficult. The necrotic hematopoietic cells gave inconclusive results on the initial immunohistochemistry tests. The prognosis of bone marrow necrosis is better secondary to acute lymphoblastic leukemia in the pediatric age group compared with adults and those with underlying solid tumors. Despite the aggressive behavior at initial presentation, the patient responded to chemotherapy and necrosis disappeared at day 28 after the start of the therapeutic regimen.

【 授权许可】

   
2012 Khoshnaw et al.; licensee BioMed Central Ltd.

【 图 表 】

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【 参考文献 】

• [1]Paydas S, Ergin M, Baslamisli F, Yavuz S, Zorludemir S, Sahin B, Bolat FA: Bone marrow necrosis: clinicopathologic analysis of 20 cases and review of the literature. Am J Hematol 2002, 70:300-305.
• [2]Eusni RM, Hamidah Hussin N, Zarina AL, Rahman J: Bone marrow necrosis preceding infantile acute lymphoblastic leukaemia. Malays J Pathol 2007, 29:113-117.
• [3]Foucar K: Miscellaneous disorders of bone marrow including stromal and bone marrow abnormalities. In Bone Marrow Pathology. 2nd edition. Chicago: ASCP Press; 2001:546-585.
• [4]Invernizzi R, D’Alessio A, Iannone AM, Pecci A, Bernuzzi S, Castello A: Bone marrow necrosis in acute lymphoblastic leukemia. Haematologica 1995, 80:572-573.
• [5]Bernard C, Sick H, Boilletot A, Oberling F: Bone marrow necrosis: acute microcirculation failure in myelomonocytic leukemia. Arch Intern Med 1978, 138:1567-1569.
• [6]Tang YM, Jeavons S, Stuckey S, Middleton H, Gill D: MRI features of bone marrow necrosis. AJR Am J Roentgenol 2007, 188:509-514.
• [7]Ranaghan L, Morris TC, Desai ZR, Markey GM: Bone marrow necrosis. Am J Hematol 1994, 47:225-228.
• [8]Norgard MJ, Carpenter JT Jr: Conrad ME: Bone marrow necrosis and degeneration. Arch Intern Med 1979, 139:905-911.
• [9]Cowan JD, Rubin RN, Kies MS, Cerezo L: Bone marrow necrosis. Cancer 1980, 46:2168-2171.
• [10]Conrad ME, Carpenter JT: Bone marrow necrosis. Am J Hematol 1979, 7:181-189.
• [11]Brown CH: Bone marrow necrosis: a study of seventy cases. Johns Hopkins Med J 1972, 131:189-203.
• [12]Kiraly JF: Wheby MS 3rd: Bone marrow necrosis. Am J Med 1976, 60:361-368.
• [13]Janssens AM, Offner FC, Van Hove WZ: Bone marrow necrosis. Cancer 2000, 88:1769-1780.
• [14]Dunn P, Shih LY, Liaw SJ, Sun CF: Bone marrow necrosis in 38 adult cancer patients. J Formos Med Assoc 1993, 92:1107-1110.
• [15]Delgado P, Giraldo P, Roca M: Alvarez R: [Magnetic resonance imaging in the early diagnosis of bone marrow necrosis] [in Spanish]. Sangre (Barc) 1999, 44:65-69.
• [16]Lee JL, Lee JH, Kim MK, Cho HS, Bae YK, Cho KH, Bae SH, Ryoo HM, Lee KH, Hyun MS: A case of bone marrow necrosis with thrombotic thrombocytopenic purpura as a manifestation of occult colon cancer. Jpn J Clin Oncol 2004, 34:476-480.
• [17]Bulvik S, Aronson I, Ress S, Jacobs P: Extensive bone marrow necrosis associated with antiphospholipid antibodies. Am J Med 1995, 98:572-574.
• [18]Lackner H, Strenger V, Sovinz P, Beham-Schmid C, Pilhatsch A, Benesch M, Schwinger W, Ulreich R, Schmidt S, Urban C: Bone marrow necrosis in a girl with Hodgkin’s disease. Support Care Cancer 2012, 20:2231-2234.
• [19]Petrella T, Bailly F, Mugneret F, Caillot D, Chavanet P, Guy H, Solary E, Waldner A, Devilliers E, Carli PM, Michiels R: Bone marrow necrosis and human parvovirus associated infection preceding an Ph1+ acute lymphoblastic leukemia. Leuk Lymphoma 1992, 8:415-419.
• [20]Aboulafia DM, Demirer T: Fatal bone marrow necrosis following fludarabine administration in a patient with indolent lymphoma. Leuk Lymphoma 1995, 19:181-184.
• [21]Limentani SA, Pretell JO, Potter D, DuBois JS, Daoust PR, Spieler PS, Miller KB: Bone marrow necrosis in two patients with acute promyelocytic leukemia during treatment with all-trans retinoic acid. Am J Hematol 1994, 47:50-55.
• [22]Kumakura S, Ishikura H, Kobayashi S: Bone marrow necrosis and the Lambert-Eaton syndrome associated with interferon α treatment. N Engl J Med 1998, 338:199-200.
• [23]Macfarlane SD, Tauro GP: Acute lymphocytic leukemia in children presenting with bone marrow necrosis. Am J Hematol 1986, 22:341-346.
• [24]Graham ML, Estrada J, Ragab AH, Starling KA, Rosen D, Wilkenson RW: Phase II trial of mitoxantrone in acute lymphocytic leukemia of childhood. A Pediatric Oncology Group study. Invest New Drugs 1991, 9:263-267.
• [25]Montemayor-Montoya JL, De León-Cantú RE, Gómez-Almaguer D, Herrera-Garza JL: 3 cases of bone marrow necrosis in acute leukemia] [in Spanish. Rev Invest Clin 1997, 49:295-298.
• [26]Pullen DJ, Boyett JM, Crist WM, Falletta JM, Roper M, Dowell B, Van Eys J, Jackson JF, Humphrey GB, Metzgar RS, Cooper MD: Pediatric oncology group utilization of immunologic markers in the designation of acute lymphocytic leukemia subgroups: influence on treatment response. Ann N Y Acad Sci 1984, 428:26-48.
• [27]Matsuzaki A, Okamura J, Ishii E, Ikuno Y, Koga H, Eguchi H, Yanai F, Inada H, Nibu K, Hara T, Take H, Miyazaki S, Tasaka H: Treatment of standard-risk acute lymphoblastic leukemia in children: the results of protocol AL841 from the Kyushu-Yamaguchi Children’s Cancer Study Group in Japan. Pediatr Hematol Oncol 1999, 16:187-199.
• [28]Ishii E, Hara T, Ohkubo K, Matsuzaki A, Takeuchi T, Ueda K: Treatment of childhood acute lymphoblastic leukemia with intermediate-dose cytosine arabinoside and adriamycin. Med Pediatr Oncol 1986, 14:73-77.