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Journal of Clinical Oncology, Vol 23, No 18 (June 20), 2005: pp. 4225-4226 © 2005 American Society of Clinical Oncology. DOI: 10.1200/JCO.2005.04.133
CNS Manifestations of MalignanciesCASE 1. Conjunctival Relapse of Acute Lymphoblastic Leukemia Heralding Pituitary and CNS DiseaseDepartments of Ophthalmology and Visual Sciences, Prince of Wales Hospital, Hong Kong
Department of Pathology, Queen Mary Hospital, Hong Kong
Medicine, Queen Mary Hospital, Hong Kong A 64-year-old man presented with acute lymphoblastic leukemia (ALL) and achieved complete remission with chemotherapy (doxorubicin, vincristine, prednisolone, and cyclophosphamide). He completed consolidation therapy with cranial radiotherapy, intrathecal methotrexate (MTX 12 mg x 6). 1 One year later, while on oral maintenance, he presented with bilateral blurring of vision (visual acuity, 0.2), generalized bone pain, and weight loss. Physical examination showed bilateral lower conjunctiva fleshy "salmon patch" infiltrates, which were biopsied (Fig 1A, arrows). Slit lamp and fundoscopic examinations were normal. A complete blood picture was normal, but a bone marrow biopsy and lumbar puncture showed blast cells. This was followed by sudden development of diabetes insipidus (daily urine output, 6 L) and panhypopituitarism (cortisol, 16nmol/L; thyroxine 7.2 pmol/L; thyroid-stimulating hormone, follicle-stimulating hormone, leutenizing hormone, adrenocorticotropic hormone, all undetectable). He was treated with intrathecal MTX (12 mg) and systemic MOD chemotherapy (MTX, 1g; vincristine, 2 mg; and dexamethasone, 40 mg). 2 This resulted in overnight resolution of the conjunctival infiltrate (Fig 1B) and bilateral visual recovery (acuity, 0.9). The conjunctival histology showed extensive subconjunctival lymphoblast infiltration (Fig 1C, arrows). Magnetic imaging scanning showed leukemic infiltration of the pituitary sella and clivus, abutting the optic chiasm (Fig 1D arrow), together with deposits in the spleen and right kidney. Three more courses of intrathecal MTX and MOD were administered, with pituitary irradiation and thyroxine, cortisol, and desmopressin acetate replacement, and the patient achieved a good partial remission.
Conjunctival relapse of ALL is reported rarely for both pediatric 3 and adult patients. 4 Most reported cases presented as isolated asymptomatic masses and were treated with radiotherapy. This is the first description of concomitant conjunctival, cerebral and pituitary relapse of ALL. Anatomically, the striking conjunctival infiltrate is unlikely to cause the visual impairment. Rather, the CSF and pituitary disease suggested direct ALL involvement of the optic nerve and tracts. Visual complaints in ALL patients often herald CNS disease and required urgent lumbar puncture and imaging. Compared with conjunctival radiotherapy, the use of systemic chemotherapy carried the advantage of speed and ease of administration, and also covers inevitable systemic disease. Despite extensive disease, our case showed good symptomatic response. The choice of MTX, which has therapeutic CSF 5 and eye globe 6 penetrating ability, may also be of benefit. Authors' Disclosures of Potential Conflicts of Interest The authors indicated no potential conflicts of interest.
REFERENCES 1. Liang R, Todd D, Chan TK, et al: Intensive chemotherapy for adult lymphoblastic lymphomas. Cancer Chemother Pharmacol 29:80-82, 1991[Medline] 2. Wiernik PH, Dutcher JP, Paietta E, et al: Long-term follow-up of treatment and potential cure of adult acute lymphocytic leukemia with MOAD: A non-anthracycline containing regimen. Leukemia 7:1236-1241, 1993[Medline] 3. Campagnoli MF, Parodi E, Linari A, et al: Conjunctival mass: An unusual presentation of acute lymphoblastic leukemia relapse in childhood. J Pediatr 142:211, 2003[Medline] 4. Cook BE Jr, Bartley GB: Acute lymphoblastic leukemia manifesting in an adult as a conjunctival mass. Am J Ophthalmol 124:104-105, 1997[Medline] 5. Hill FG, Richards S, Gibson B, et al: Successful treatment without cranial radiotherapy of children receiving intensified chemotherapy for acute lymphoblastic leukaemia: Results of the risk-stratified randomized central nervous system treatment trial MRC UKALL XI (ISRC TN 16757172). Br J Haematol 124:33-46, 2004[CrossRef][Medline]
6. Batchelor TT, Kolak G, Ciordia R, et al: High-dose methotrexate for intraocular lymphoma. Clin Cancer Res 9:711-715, 2003
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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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