Bone Marrow Transplantation (2009) 44, 131–132 & 2009 Macmillan Publishers Limited All rights reserved 0268-3369/09 $32.00 www.nature.com/bmt

LETTER TO THE EDITOR Diphyllobothriasis, a rare cause of profuse diarrhea following autologous transplantation

Bone Marrow Transplantation (2009) 44, 131–132; involved areas. Biopsyof the right cervical lymphnode doi:10.1038/bmt.2008.439; published online 26 January2009 confirmed relapse of anaplastic T-cell lymphoma. A microbiologyconsultation was obtained on day þ 42; three stool specimens were sent for parasitic examination. A diagnosis of D. latum infection was made after Diphyllobothriasis is a zoonosis acquired by humans after identification of numerous characteristic parasite eggs the ingestion of plerocercoid larvae present in raw and (Figure 2). The patient received a single dose of prazi- undercooked fish. In North America, Diphyllobothrium quantel (10 mg/kg) on day þ 43 with initial improvement latum (D. latum) infection is limited mostlyto areas in stool output. Two stool specimens became negative with cold water lakes.1 Various species of freshwater or for parasite eggs after the first treatment. However, on anadromous (living in both fresh and saltwater) fishes may day þ 49, profuse diarrhea recurred and eggs in two separate be infected by D. latum plerocercoid larvae, including stool specimens were again identified. A second dose of perch, trout, salmon, char and pike.2 We report herein the first case of D. latum infection in an immunocompromised patient who presented with profuse diarrhea. 6000 The patient was a 60-year-old man who lived in the north-east area of the Province of Quebec, Canada, and who had never traveled outside the country. This patient 5000 with anaplastic T-cell lymphoma had achieved minimal residual disease with ESHAP (etoposide, methylpredniso- 4000 lone, cytosine arabinoside and cis-platinum) chemotherapy. He was admitted to our institution in December 2005 to undergo auto-SCT. His disease initiallypresented with 3000 widespread abdominal lymph nodes and multiple hepatic nodular lesions, which progressed after eight cycles of CHOP (CY, doxorubicin, VCR and prednisone). His 2000 medical historywas significant for mild, chronic diarrhea (2–3 loose stools/day) of 1-year duration; complete investigation bya gastroenterologist, including upper and 1000 lower endoscopies, was negative. After conditioning with BEAC (bis-chloronitrosourea, 0 etoposide, cytosine arabinoside and CY), the patient received his autologous stem cell graft on 29 December 110192837465564 2005. On day þ 3, though neutropenic, he developed fever, Day after autologous transplant diffuse abdominal pain and profuse diarrhea, which Figure 1 Evolution of stool output in ml after autologous transplant. reached a maximum of 5025 ml/day(Figure 1). A computed Arrows indicate Praziquantel treatment. tomographyscan of the abdomen revealed right colitis as well as a thickening of the cecum and distal parts of the small bowel. Coprological cultures were negative for pathogenic bacteria or viruses; quantitative PCR in blood leukocytes for CMV was also negative. The patient recovered from aplasia on day þ 18 with decreased diarrhea. However, the improvement was of short duration, with recurrence of both diarrhea and fever on day þ 28 despite the resolution of aplasia (Figure 1). Multiple repeat stool cultures were unable to identifyanyspecific pathogen. A careful physical examination showed a new right cervical lymph node. Repeat computed tomography scans of the chest and abdomen were suggestive of lymphoma relapse Figure 2 Eggs of Diphyllobothrium latum found in the feces of the with recurrence of several lymph nodes in previously patient (Microscopy  10). Letter to the Editor 132 praziquantel on day þ 57 led to complete resolution of antibiotics have been incriminated. The impact of multiple diarrhea; all stool examinations remained negative after the concomitant medications should also be considered. In our second dose. Owing to earlyrelapse after transplant, the patient, the stool output was much greater than what is patient was offered palliative care and died on day þ 82. usuallyexpected following a BEAC conditioning regimen. This is the first report of D. latum infection in an In addition, diarrhea recurred without the introduction immunocompromised host. The major symptoms encoun- of anynew therapeutic agent, a finding dismissing a tered by D. latum infection in immunocompetent patients drug-related side effect. include abdominal pain, vomiting, dyspepsia, diarrhea and In summary, we report the first case of D. latum infection discharge of proglottids in feces.3 Patients often seek in an immunocompromised host. This report is of medical advice after observing tapeworm segments in feces. particular importance in view of the widespread increase Other symptoms are nonspecific and include fatigue, in consumption of raw fish, especiallysushis or fish tartare, anorexia, fever and myalgia.3,4 Megaloblastic anemia has in developed countries. Search for parasites should clearly been described as a complication of long-term infestation, be a part of stool examinations in patients who develop as a result of vitamin B12 malabsorption.5 In our patient, it diarrhea after transplant. is difficult to determine whether the initial symptoms (fever, M Detrait1, L Poirier2, D-C Roy1, T Kiss1, L Busque1, abdominal pain) before autologous transplant were due S Cohen1, S Lachance1, G Sauvageau1 and J Roy1 to lymphoma or parasitic infection. There was no evidence 1Blood and Marrow Transplant Program, of eosinophilia, megaloblastic anemia or vitamin B12 Maisonneuve-Rosemont Hospital, University of Montreal, deficiencyin blood analysesbefore transplant. Following Montreal, Quebec, Canada and further questioning on life habits, we discovered that the 2Department of Clinical Microbiology, most likelysource of contamination was the consumption Maisonneuve-Rosemont Hospital, University of Montreal, of raw trout in the months before autograft. Montreal, Quebec, Canada Since 1987, the medication of choice for D. latum E-mail: [email protected] infection has been praziquantel. Single-dose treatment with praziquantel 5–10 mg/kg produces high cure rates in patients with D. latum infection. However, in many instances, a second dose is given when the tapeworm is References not evacuated. In our patient, a second dose was given after the recurrence of diarrhea and relapse of eggs on stool 1 Dick TA, Nelson PA, ChoudhuryA. Diphyllobothriasis:update examination. It is noted that the whole worm was not on human cases, foci, patterns and sources of human infections observed in feces after the first praziquantel dose.6,7 and future considerations. Southeast Asian J Trop Med Publ Diarrhea is often observed after autologous or allogeneic Health 2001; 32 (Suppl 2): 59–76. transplantation. Identification of the specific cause is 2 Dupouy-Camet J, Peduzzi R. Current situation of human important for appropriate management. Diarrhea may diphyllobothriasis in Europe. Eurosurveillance 2004; 9: 31–35. have multiple causes, including infectious agents, drugs, 3 Lee EB, Song JH, Park NS, Kang BK, Lee HS, Han YJ et al. GVHD and metabolic conditions. Among infectious A case of Diphyllobothrium latum infection with a brief review of diphyllobothriasis in the Republic of Korea. Korean agents, the bacterial, viral and parasitic organisms can all J Parasitol 2007; 45: 219–223. cause diarrhea. In the first 30 days after transplantation, 4 Tanowitz HB, Weiss LM, Wittner M. Tapeworms. Curr Infect 8 Clostridium difficile is the most common pathogen. Dis Rep 2001; 3: 77–84. After 30 days, CMV disease predominates. Herpes virus, 5 Vuylsteke P, Bertrand C, Verhoef GE, Vandenberghe P. Case of enterovirus and rotavirus have also been reported. Crypto- megaloblastic anemia caused byintestinal taeniasis. Ann sporidium spp, Entamoeba histolytica and Blastocystis Hematol 2004; 83: 487–488. hominis are parasites most commonlyseen in immunocom- 6 Groll E. Praziquantel for cestode infections in man. Acta Trop promised patients. Cryptosporidiosis has been reported, 1980; 37: 293–296. especiallyin renal transplant recipients. Strongyloides and 7 Tanowitz HB, Weiss LM, Wittner M. Diagnosis and treat- Ascaris are rare, whereas Mycobacterium avium and ment of intestinal helminthes. I. Common intestinal cestodes. Gastroenterologist 1993; 1: 265–273. Candida albicans are uncommon etiological agents of 8 Forrest G. Gastrointestinal infections in immunocompromised 8,9 diarrhea. hosts. Curr Opin Gastroenterol 2004; 20: 16–21. Drug-induced diarrhea also occurs frequentlyamong 9 Helderman JH. Prophylaxis and treatment of gastrointestinal transplant patients. Both chemotherapeutic agents, such as complications following transplantation. Clin Transplant 2001; melphalan, thiotepa, anthracyclines and etoposide, and 15: 29–35.

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