Granulomatous pneumonitis following bone marrow transplantation

Abstract

Summary:We describe the rare occurrence of a granulomatous pneumonitis seen in a patient following allogeneic bone marrow transplantation. Interestingly sarcoidosis was diagnosed in the marrow donor less than a year after donating his bone marrow. Bone Marrow Transplantation (2001) 28, 627-630. Keywords: alveolitis; extrinsic allergic; sarcoidosis; bone marrow transplantation; granuloma Case report, recipientA 37-year-old white female presented with a lump in her neck in November 1996. Biopsy revealed a large B cell lymphoma. Chest and abdominal CT scans showed axillary, mediastinal and retroperitoneal adenopathy and a solitary left pulmonary nodule. Bone marrow examination was negative for lymphomatous involvement. She received six cycles of chemotherapy comprising cyclophosphamide, adriamycin (total dose 300 mg/m 2 ), procarbazine and prednisone. Follow-up evaluations showed progression of disease with an increase in axillary adenopathy and the appearance of bilateral pulmonary nodules. She was then given salvage chemotherapy with etoposide, methylprednisolone, cisplatinum and cytarabine.She underwent an HLA-identical bone marrow transplant from her brother in June 1997. The HLA type was A (1,26); B(8,49); Bw(4,6); Cw (7,-); DR␤1‫;11030ء‬ DR␤1‫.2031ء‬ The conditioning regimen consisted of VP-16, 60 mg/kg and TBI dosing of 225 cGy for 6 days. She received methotrexate and cyclosporine for graft-versushost disease (GVHD) prophylaxis and engrafted on day 13 following the transplantation. She developed a skin rash 140 days following the transplant and was treated for cutaneous GVHD with prednisone and cyclosporine that were tapered off in July 1998. A chest X-ray in June 1998 showed a small ill-defined opacity in the right lower

Sarcoidosis as an unusual cause of hepatic dysfunction following reduced intensity conditioned allogeneic stem cell transplantation

“…Of interest is that three of the four previously reported patients received stem cells from siblings diagnosed with sarcoidosis. 5,7,8 However, in our patients neither of the donors had any evidence of sarcoidosis, and it is possible that the immune dysregulation following alemtuzumab-based conditioning 10 may have precipitated the development of sarcoidosis.…”

“…6 In contrast, it is rare to encounter sarcoidosis of the liver after HSCT. 5 We know of only four previous cases of patients developing sarcoidosis following allogeneic stem cell transplantation, 5,7-9 and of only one with hepatic sarcoidosis. 7 All previously reported patients received standard conditioning regimens, whereas both our patients underwent RIC-HSCT.…”

“…3 Sarcoidosis has been associated with infections, inflammatory conditions, and/or drugs, but it has rarely been reported following haemopoietic stem cell transplantation (HSCT). 4,5 We report two patients who developed liver sarcoidosis following a reduced intensity conditioned (RIC) allogeneic HSCT.…”

Evidence for Mycobacteria in Sarcoidosis

“…In case-control studies from the Isle of Man, 40% of sarcoidosis cases reported contact with a patient with sarcoidosis before disease onset, compared with only 1% of control subjects (25,26). Finally, some patients developed sarcoidosis after receiving a transplant from a patient with sarcoidosis (27)(28)(29)(30). These observations strongly support the presence of a transmissible agent in sarcoidosis.…”

Donor cell-derived sarcoidosis after allogeneic BMT

“…[1][2][3][4][5] Previous study never referred to chimerism of sarcoidosis lesions. This is the first report in which chimerism of lymph nodes where sarcoidosis had infiltrated was investigated and the first study showing that lymph nodes contained mostly donor-type cells.…”