Journal List > J Rheum Dis > v.24(4) > 1064335

Ryu, Park, Sheen, Kim, Han, Jung, Suh, and Hwang: Cytoplasmic Anti-Neutrophil Cytoplasmic Antibody Positive Diffuse Alveolar Hemorrhage Associated with Methimazole

Abstract

Diffuse alveolar hemorrhage (DAH) is a life-threatening condition associated with many disorders. Here, we report a case of 59-year-old female who had diffuse alveolar hemorrhage associated with methimazole. She had been treated with methimazole for two weeks due to the recurrence of Grave's disease, before visiting the emergency room. She had to be intubated on the 3rd day of hospitalization because of unabated massive hemoptysis and rapid progression of diffuse alveolar infiltration on chest radiographs. Since her clinical condition improved substantially after cessation of methimazole and steroid pulse therapy, she was extubated on the 9th day of hospitalization and then discharged. After discharge, DAH did not recur with cessation of steroid and she had radioactive iodine therapy for her Grave's disease. This was a rare and interesting case of life-threatening DAH associated with cytoplasmic-antineutrophil cytoplasmic antibody and methimazole.

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Figure 1.
Diffuse consolidation and ground glass opacity are observed on both lungs of initial chest X-ray (A). Initial chest computed tomography shows bilateral involvement of diffuse ground glass opacity, interlobular septal thickening, and multifocal consolidations (B).
jrd-24-236f1.tif
Figure 2.
Chest X-rays at intensive care unit showed gradual improvement. On hospital day 3 mechanical ventilator care and steroid pulse therapy were started (A). On hospital day 5, both lung consolidations were improved (B). On hospital day 9, extubation was done (C). On hospital day 13, the patient was discharged from the hospital (D).
jrd-24-236f2.tif
Figure 3.
Bronchoscopy (A: main carina, B: left, second car-ina, C: right upper lobe branch) performed on hospital day 5 showed no endobronchial lesion.
jrd-24-236f3.tif
Table 1.
Summary of cases of diffuse alveolar hemorrhage with MPO-ANCA associated with anti-thyroid drugs
Drug Race Gender/ Age (yr) Other feature Anti-MPO Anti-PR3 Tx Outcome Reference
MMI Korean F/59 -   + S Improved Present case
PTU Caucasian F/27 Athralgia, +   SP Improved Ortiz-Diaz et al.
      renal failure          
PTU Japanese F/69 - +   S Improved Wakabayashi et al.
PTU/MMI Chineses F/44 Hematuria +   S Improved Lau et al. [6]
PTU Korean Pregnant F/25 GN +   S Improved Kang et al.
CMZ Spanish F/52 GN +   SC Improved Calañas-Continente et al.
PTU NA F/12 - +   S Improved Pirot et al.
PTU Japanese F/54 Hematuria +   S Improved Nakamori et al.
PTU Japanese F/59 - + + - Improved Yamauchi et al.
PTU Japanese F/66 Hematuria +   S Improved Katayama et al. [11]
PTU Caucasian F/33 - +   SC Died Seligman et al.
MMI Chinese F/18 Skin, nerve +   SP Improved Tsai et al. [10]
PTU NA Pregnant F/23 GN +   SP Improved Dhillon et al.
PTU Japanese F/44 Hematuria +   - Improved Ohtsuka et al.
PTU NA F/39 Arthritis +   - Improved Romas et al.

MPO: myeloperoxidase, ANCA: anti-neutrophil cytoplasmic antibody, PR3: proteinase 3, Tx: treatment, MMI: methimazole, PTU: propylthiouracil, CMZ: carbimizole, F: female, GN: glomerulonephritis, S: steroid, P: plasmapheresis, C: cyclophosphamide, NA: not available.

* Extra references for Table 1.

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Kang AY, et al. Korean J Intern Med 2006;21:240-3. Nakamori Y, et al. Intern Med 2003;42:529-33.

Ohtsuka M, et al. Eur Respir J 1997;10:1405-7. Ortiz-Diaz EO. BMJ Case Rep 2014 Aug 22 [Epub] DOI: 10.1136/bcr-2014-204915.

Pirot AL, et al. Pediatr Pulmonol 2005;39:88-92. Romas E, et al. J Rheumatol 1995;22:803.

Seligman VA, et al. J Clin Rheumatol 2001;7:170-4.

Wakabayashi K, et al. Nihon Ronen Igakkai Zasshi 2012;49:612-6. Yamauchi K, et al. Respirology 2003;8:532-5.

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