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Journal of Clinical Oncology, Vol 23, No 6 (February 20), 2005: pp. 1314-1315 © 2005 American Society of Clinical Oncology. DOI: 10.1200/JCO.2005.02.085
Uncommon Presentations of Some Common MalignanciesCASE 2. Nasopharyngeal Carcinoma Followed by Secondary Acute Promyelocytic Leukemia Presenting With Respiratory DistressDepartments of Medicine, Surgery, and Pathology, Queen Mary Hospital, University of Hong Kong, Hong Kong, Republic of China A 43-year-old Chinese man presented with recurrent epistaxis and left cervical lymph nodes. Endoscopic examination and biopsy showed nasopharyngeal carcinoma with impending base of skull erosion. He was treated with combined chemotherapy (cisplatin 60 mg weekly in four doses) and local radiotherapy (RT; 60 Gy with 16-Gy boost). He had severe mucositis without neutropenia and achieved complete remission with no neurologic deficit. Sixteen months later, he presented with fever, neck pain, and sore throat. This progressed rapidly to hoarseness, dysphasia, and nasal regurgitation. Complete blood studies showed hemoglobin, 7.0g/dL; white cell count, 3.3 x 109/L (80% promyelocytes); and platelet count, 152 x 109/L with normal coagulation. A bone marrow biopsy confirmed acute promyelocytic leukemia (APL). His condition progressed rapidly to stridor. Endoscopy followed by computerized tomogram (Figs 1A and 1B) showed severe mucosal swelling from the nasopharynx down to the epiglottis and vocal cords. An emergency cricothyroidotomy was performed for asphyxia under platelet and plasma coverage. Biopsy of the swollen tissue showed infiltration by APL blast cells with extensive accompanying tissue edema. These blast cells expressed myeloperoxidase, but were negative for CD56 (Figs 1C and 1D). Despite the administration of all-trans-retinoic acid via orogastric tube and intravenous dexamethasone and chemotherapy, he died as a result of hypoxic convulsions.
Therapy-related APL (t-APL) is increasingly recognized as a complication of previous RT and chemotherapy. In our experience, tAPL accounted for 2% of all APL and 14% of all therapy-related acute myelocytic leukemia (update from Au1). Clinically and pathologically, they are indistinguishable from de novo APL and respond similarly to treatment.2 Although patients with nasopharyngeal carcinoma have an increased risk of leukemia,3 t-APL is seldom reported and the predilection of the APL cells to the upper aerodigestive track is intriguing. We previously have reported two cases of APL relapses with external auditory canal infiltration in patients with recurrent otitis externa.4 Likewise, the APL cells in our patient case may also be attracted by subclinical local inflammation due to previous RT and mucosal damage. The aggregation of APL cells is under the regulation of specific adhesion molecules (eg, LFA-1 and ICAM-2),5 and deranged regulation of these molecules persists long after RT.6 Clinically, the acute nature and strategic site of such APL infiltration makes the delivery of surgical and medical treatment extremely difficult and hazardous. Authors' Disclosures of Potential Conflicts of Interest The authors indicated no potential conflicts of interest.
REFERENCES 1. Au WY, Ma SK, Chung LP, et al: Two cases of therapy-related acute promyelocytic leukemia (t-APL) after mantle cell lymphoma and gestational trophoblastic disease. Ann Hematol 81:659–661, 2002[Medline]
2. Beaumont M, Sanz M, Carli PM, et al: Therapy-related acute promyelocytic leukemia. J Clin Oncol 21:2123–2137, 2003 3. Wang CC, Chen ML, Hsu KH, et al: Second malignant tumors in patients with nasopharyngeal carcinoma and their association with Epstein-Barr virus. Int J Cancer 87:228–231, 2000[CrossRef][Medline]
4. Au WY, Chan GC, Chim CS, et al: Unusual sites of involvement by hematologic malignancies: Case 3. External auditory canal tumor—A rare chloroma in acute promyelocytic leukemia with a complete response to arsenic trioxide. J Clin Oncol 19:3993–3995, 2001
5. Larson RS, Brown DC, Sklar LA: Retinoic acid induces aggregation of the acute promyelocytic leukemia cell line NB-4 by utilization of LFA-1 and ICAM-2. Blood 90:2747–2756, 1997 6. Prott FJ, Handschel J, Micke O, et al: Long-term alterations of oral mucosa in radiotherapy patients. Int J Radiat Oncol Biol Phys 54:203–210, 2002[Medline]
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Copyright © 2005 by the American Society of Clinical Oncology, Online ISSN: 1527-7755. Print ISSN: 0732-183X
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