CASE REPORT Pulmonary actinomycosis and Hodgkin s disease ... · PDF fileCASE REPORT Pulmonary...

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  • CASE REPORT

    Pulmonary actinomycosis and Hodgkins disease:when FDG-PET may be misleadingC Weisshaupt,1 F Hitz,1 W C Albrich,2 A Omlin1

    1Department of Oncology/Hematology, KantonsspitalSt. Gallen, St. Gallen,Switzerland2Department of Infectiology,Kantonsspital St. Gallen,St. Gallen, Switzerland

    Correspondence toDr C Weisshaupt,christian.weisshaupt@kssg.ch

    Accepted 31 August 2014

    To cite: Weisshaupt C,Hitz F, Albrich WC, et al.BMJ Case Rep Publishedonline: [please include DayMonth Year] doi:10.1136/bcr-2014-206034

    SUMMARYWe present a patient with advanced Hodgkins diseasetreated with escalated BEACOPP chemotherapy. Theresult from the interim fluorodeoxyglucose positronemission tomography with CT (PET-CT) after two cyclesof chemotherapy is crucial for treatment guidance for theclinical trial HD18 from the German Hodgkin StudyGroup. An increase in size and standard uptake value(SUV) of a pulmonary lesion suggesting refractoryHodgkins disease was documented. Since all othermanifestations of the lymphoma responded well to thetreatment, the discordant behaviour was suspicious foranother reason for this progressive pulmonary lesion.Bronchoscopy revealed Actinomyces species in culturesfrom bronchial washings. Specific treatment was initiatedand consisted of 2 weeks of intravenous penicillinfollowed by ceftriaxone intravenous for another 4 weeksand subsequent oral amoxicillin to complete 12 monthsof antibiotic therapy. For the Hodgkins lymphoma,complete remission was documented after a total of sixcycles of escalated BEACOPP.

    BACKGROUNDHodgkins lymphoma is a common malignantdisease that, if treated with a combination of chemo-therapy and radiotherapy, has high complete remis-sion rates of 8090% even if diagnosed as advanceddisease.1 The current standard clinical staging proce-dures include positron emission tomography withCT (PET-CT) scanning.2 Furthermore, PET-CT iscurrently evaluated in large clinical trials (GermanHodgkin Trial Group, HD 18, NCT00515554) as atool for early decision-making.We present a young patient with Hodgkins

    disease whose PET-CT demonstrated improvementin some but progression in other lesions withincreased uptake on PET scan after two cycles ofdose-intense chemotherapy.Discordant behaviour of suspected tumour

    lesions in a highly chemotherapy sensitive diseasesuch as Hodgkins lymphoma can lead to uncer-tainty for the patient and the treating physician andshould raise suspicion for alternative explanationsfor PET-positive lesions.

    CASE PRESENTATIONA 38-year-old Swiss chimneysweeper with asmoking history of 12 pack years was hospitalisedfor productive cough over the last 2 months,weight loss of 10 kg during the last 6 months andnight sweats. The medical history was completelyunremarkable. In a CT scan, mediastinal lymph-adenopathy and a pleural effusion on the left side

    were seen. A PET-CT scan showed increased stand-ard uptake value (SUV) of a mediastinal mass,supraclavicular up to cervical lymph nodes on theleft side and pericardial, pleural, diffuse pulmonary(left upper lobe) and bone lesions (figure 1A).Bronchoscopy and biopsy of mediastinal lymphnodes were not conclusive but cytologically showedcells suspicious for malignant disease. Since furtherdifferentiation was not possible, Hodgkins lymph-oma, non-Hodgkins lymphoma and seminomawere included in the differential diagnosis due tolocalisation and age of the patient.The final diagnosis was established by a supracla-

    vicular lymph node excision, which revealed a clas-sical nodular-sclerosing Hodgkins lymphoma. AnnArbor stage based on symptoms and PET-CT scanwas IVBE.The reason for the productive cough was consid-

    ered to be a community-acquired pneumonia andthe patient was treated empirically with amoxicillinand clavulanic acid because of an elevated C react-ive protein and pneumonia-like clinical features,with prompt relieve of these symptoms after 7 daysof antibiotic treatment. For the Hodgkins lymph-oma the patient was enrolled in a study-protocolfrom the German Hodgkin Study Group, HD18with escalated BEACOPP (NCT00515554). Hereceived two cycles of escalated BEACOPP fol-lowed by treatment-evaluation with an interimPET-CT scan.All the lesions reported on the initial PET-CT

    showed a good metabolic response apart from thepulmonary infiltration in the apical left upper lobeof the lung, which on the new PET-CT showed pro-gressive SUV and on the CT component hadincreased in size and density (figure 1B).

    INVESTIGATIONSPrimary chemotherapy refractory Hodgkinsdisease based on the progressive lung lesion wasconsidered to be very unlikely. Therefore, differen-tial diagnoses included mainly infections, despite alack of clinically apparent signs of infection.A review of the patients charts showed a positiveculture for Actinomyces graevenitzii from the initialbronchoscopy. The culture was initially interpretedas possibly contaminated in the context of diagno-sis of a malignant lymphoma and successful treat-ment of the pulmonary symptoms with a shortcourse of antibiotics. In the light of a progressivelung-lesion on chemotherapy the diagnosis of pul-monary actinomycosis was further investigated withrepeat bronchoscopy. The cultures from the bron-chial washing revealed A. graevenitzii and A.

    Weisshaupt C, et al. BMJ Case Rep 2014. doi:10.1136/bcr-2014-206034 1

    Reminder of important clinical lesson

    http://crossmark.crossref.org/dialog/?doi=10.1136/bcr-2014-206034&domain=pdf&date_stamp=2014-09-20http://casereports.bmj.com

  • odontolyticus, which were both sensitive to penicillin and cef-triaxone. Furthermore, the initial transbronchial biopsy of aparatracheal lymph node grew Actinomyces species. Microscopyand culture of bronchial lavage and lymph nodes were negativefor other bacteria, mycobacteria and fungi. PCR forMycobacterium tuberculosis was negative from bronchoalveolarlavage and lymph node.

    DIFFERENTIAL DIAGNOSISDifferential diagnosis included various causes of lung infectionssuch as tuberculosis, aspergillosis, nocardiosis, actinomycosis orindeed primary refractory Hodgkins disease of the lung. Thepatient received prophylactic antibiotic treatment withcotrimoxazole as per protocol of the HD18 trial.

    TREATMENTInitially the patient received penicillin 4 Mio IU intravenouslyevery 4 h for 2 weeks. To allow subsequent outpatient manage-ment the antibiotic treatment was switched after 2 weeks to cef-triaxone (2 g intravenously every 24 h) for a total of 4 weeks.A repeat high-resolution CT scan of the chest 6 weeks after initi-ation of antibiotic treatment showed subtotal regression of theinfiltrations in the left upper lobe and treatment was changed tooral amoxicillin (1 g every 8 h) for a total antibiotic treatmentduration of 12 months.

    The chemotherapy was completed without complications fora total of six cycles of escalated BEACOPP as per HD18protocol.

    OUTCOME AND FOLLOW-UPThe PET-CT scan 4 weeks after completion of six cycles of esca-lated BEACOPP for stage IV Hodgkins disease and a total of3 months of antibiotic treatment for pulmonary actinomycosisshowed a complete metabolic response for the lymphoma andalmost complete resolution of the infiltration in the left upperlobe of the lung (figure 1C).

    DISCUSSIONPulmonary actinomycosis is a rare infectious disease caused byactinomyces species, mostly by Actinomyces israelii, but otherpathogenic species have been reported, such as in our caseA. graevenitzii and A. odontolyticus. Actinomyces species aremicroaerophilic or anaerobic Gram-positive rods; histologically,sulfur granules are typically found.3 Actinomycetes are normalinhabitants of the oral cavity or female genital tract. Growth insputum or bronchial secretions is considered no proof of activedisease, while the diagnosis of (pulmonary) actinomycosisrequires growth from a sterile specimen, demonstration ofsulfur granules from pus or from a histological specimen.Characteristically, actinomycosis does not respect natural bound-aries and can therefore be confused with malignancy.Oral-cervicofacial disease is the most frequent clinical manifest-ation of actinomycosis. Pulmonary infection accounts for about15% of all actinomycotic infections.4 Clinical presentationsinclude history of productive cough, haemoptysis, fever, weightloss and chest pain. Predisposing factors for pulmonary actino-mycosis include poor oral hygiene with aspirations and struc-tural underlying lung disease namely a history of tuberculosis orbronchiectasis. Radiological findings are generally unspecific:the most common findings on CT scans are consolidations(74.5%), followed by hilar lymph node enlargement (29.8%),atelectasis (28.7%), cavitation (23.4%) and ground-glass opacity(14.9%).5

    Our patient, with a concurrent diagnosis of pulmonary acti-nomcycosis and stage IVBE Hodgkins disease, had classicalsymptoms for both diseases. The productive cough and feverwere interpreted as non-specific pulmonary infection due to theextensive stage of the Hodgkins disease with pulmonary,pleural and pericardial infiltration. Despite the smoking historythere was no underlying lung disease in our patient and oral anddental hygiene were good.

    Our patient had a small consolidation in the left upper lungwith a slight FDG-uptake at first presentation. FDG-uptake and

    Figure 1 (A) CT and positron emission tomography (PET) before treatment including pathological mediastinal lymph nodes; (B) CT and PET after2 months of treatment; (C) CT and PET 4 weeks after treatment of the lymphoma and after 3 months of antibiotic treatment of the pulmonaryactinomycosis.

    2 Weisshaupt C, et al. BMJ Case Rep 2014. doi:10.1136/bcr-2014-206034

    Reminder of important clinical lesson

  • dimensions increased during intensive chemotherapy forHodgkins disease, possibly as a result of immunosuppression. Acase of a disseminated pulm