A “mysterious” intrabdominal mass with infectious origin, in a patient with HIV infection under control. Clinical Management Issues A “delayed diagnosis” allows Case report to enlarge our knowledge, by assessing a rare disease Sergio Sabbatani 1, Roberto Manfredi 1, Benedetta Fabbrizio 2, Antonio Caira 3, Fabio Filippo Trapani 1, Giovanni Fasulo 1, Pierluigi Viale 1 Abstract A probable case report of an abdominal botryomycosis has been hypothesized in a patient with a stable HIV infection under an effective antiretroviral therapy. Hyperpyrexia, abdominal pain and tenderness, and a thickening of small intestinal walls associated with multiple mesenteric adenopathies and a peritoneal involvement, prompted an ultrasonography-guided fine needle biopsy, and later a laparoscopy-laparotomy which excluded a neoplastic or lymphoproliferative disorders, showing only abundant fibrotic and necrotic-steatonecrotic tissue, with sparse multinuclear giant cells type Langhans. The prompt response to surgical intervention and a treatment with i.v. meropenem alone might be referred to a concurrent gram-negative infection 1 Department of Infectious of abdominal origin, until a late culture of an atypical Mycobacterium came to our attention Diseases, University over one month after the end of hospitalization. An updated literature search is presented and of Bologna, S. Orsola- Malpighi Hospital, discussed, in relationship with the observed, extremely infrequent case reports of botryomycosis Bologna, Italy in different clinical settings. 2 Department of Pathology and Histopathology, University of Bologna, S. Keywords: Intrabdominal mass; Peritoneal involvement; Inflammatory signs; Surgical Orsola-Malpighi Hospital, treatment; Meropenem; Botryomycosis; Atypical mycobacteriosis Bologna, Italy Una “misteriosa” massa intraddominale a eziologia infettiva, in un paziente con infezione da 3 Department of HIV controllata. Un “ritardo diagnostico” consente di approfondirne la conoscenza studiando Surgery and Organ Transplantation, University una patologia rara of Bologna, S. Orsola- CMI 2011; 5(3): 95-106 Malpighi Hospital, Bologna, Italy INTRODUCTION the final microbiological diagnosis to the “Discussion” section, we aim to leave some An organizative hitch characterized by an unintentional delayed communication, did not affect the positive clinical evolution of a patient, and allowed us to study in depth Why do we describe this case an atypical clinical case in terms of differ- The modern medicine makes use of sensi- ential diagnosis. A literature search and the tive and specific laboratories technologies, discussion among all clinicians which come which allow to make important diagnosis from this clinical presentation enabled us in short periods of time. But sometimes to contribute with personal, professional this isn’t true. The late availability of a knowledge of every specialist, and may rep- microbiological specimen has allowed to Corresponding author resent a stimulating subject for a debate also establish the clinical features by the defini- Dott. Roberto Manfredi for readers. Only after writing down this Infectious Diseases, University tive diagnosis of atypical mycobacteriosis. of Bologna, S. Orsola Hospital contribution, we were finally informed of The treatment for a long period with only Via Massarenti 11 the exact microbiological diagnosis, so that one carbapenem antibiotic did not affect I-40138 Bologna, Italy we voluntary introduced this short premise. Telephone: +39-051-6363355 the clinical response of the patient Telefax: +39-051-343500 When postponing the communication of [email protected] ©SEEd Tutti i diritti riservati Clinical Management Issues 2011; 5(3) 95 An intrabdominal-peritoneal mass during HIV infection time and some “suspense” to the readers too, nutrition, alcoholism, HIV infection, major in order to underline the adjunctive diagnos- or minor trauma, a chronic granulomatous tic difficulties potentially descending from disease, and prior surgery [2,8,10-16]. apparently lacking laboratory data in an Also the pathogenesis of botryomyco- extremely complicated diagnostic “puzzle”, sis is not completely known: the process is and the need to always maintain an elevated, thought to involve a combination of sup- broad spectrum mind in the clinical manage- porting factors including an inciting event ment of “difficult to treat” patients. (i.e. a major or minor trauma, including Botryomycosis has been described since piercing for example), the amount of inocu- 1950s as an uncommon bacterial infection lated microorganisms, the intrinsic virulence mimicking actinomycosis and fungal in- of infecting pathogens, and the intrinsic host fections, characterized by one or multiple susceptibility [1-3,6,16]. aspecific suppurative-granulomatous foci Since its first report in humans published containing sulphur-like granules, usually in 1913 [17], botryomycosis remained dif- with eosinophilic infiltrates, where in many ficult to distinguish from actinomycosis cases either Gram-positive organisms (i.e. and fungal diseases, in both cutaneous and Staphylococcus aureus, coagulase-negative visceral localizations. When the respira- Staphylococci, Streptococcus spp., Bacillus or tory tract is involved, actimomycosis usually Corynebacterium spp.), or Gram-negative has an aspiration origin, while the factors organisms (i.e. Escherichia coli, Pseudomonas prompting botryomycosis have not been aeruginosa, Proteus or Neisseria spp.), and also identified yet, with host factors and foreign anaerobe bacteria (including Actinobacillus bodies probably playing some role in its and Peptostreptococcus spp., and Propionibac- pathogenesis [1-3,7,18,19]. terium acnes), might be cultured: sometimes a A retrospective, historical re-appraisal of mixed bacterial flora may be found [1-4]. botryomycosis, may be found in the nar- Actually, after the early observations car- ration of the Philoctetes’s diseases by So- ried out in animals (especially cattle and phocles masterpiece [20,21], with reference horses), the term “botryomycosis” has been to the long-term granulomatous, non-heal- proposed by Rivolta in 1884 [1,5], after ing cutaneous wounds of the Greek hero noticing the “grapelike” appearance of its Philoctete, which occurred after a painful macroscopic lesions, which resembled those but not lethal snake (viper) bite at his foot. caused by fungi (hence the suffix “mycosis”). The superinfection of this lesion caused the Later, Magrou proved the most common legendary, very prolonged stay at the isle of bacterial origin of botryomycosis, by isolat- Lesmos of the Greek hero, where Philoctete ing S. aureus from pulmonary lesions [6], was reclaimed by his companions in order to and also demonstrated that the unusual prompt a positive course to the long-lasting histopathological picture of botryomycosis Troy war [20-22]. The limb lesion of Phi- was the result of a sort of “symbiotic” rela- loctete was described as a painful and ex- tionship between the inoculum microor- tremely chronic ulcer, not lethal in its course ganism dose, the virulence of the different but still present after around one decade, and pathogens, and the immune response of the complicated by bleeding and a discharge of affected host [1,6]. malodorous and purulent material, so that Although primarily considered as a vet- it caused severe functional impotence. Some erinary disease, over one hundred of human Homer’s commenters interpreted the lesion cases have been described in the past century, of Philoctete as caused by maduromycosis, in form of single reports or small case series. mycetoma, chromoblastomycosis, and also The majority of described episodes involved botryomycosis. A comparison between the mainly skin and skin structures [2,7], and description of the clinical features of Phi- more infrequently the thorax and the abdo- loctetes’s disease and that of very similar af- men (the so-called visceral botryomycosis, flictions (also called actinophytosis, or bac- which remains a rare disease, often described terial pseudomycosis, pyogenic granuloma, in the compromised host, although the spe- or granular bacteriosis, in some narrations) cific role of host immune response in the [1,23] shows a clinical resemblance of bot- pathogenesis of visceral botryomycosis is not ryomycosis, since each of the considered dis- fully understood) [2,3,8,9]. Possible adjunc- eases has a chronic course, may frequently tive host risk factors associated with both affect the extremities, may be caused by an cutaneous and visceral botryomycosis in- initial trauma, may present with ulcers, and clude: diabetes mellitus, cystic fibrosis, mal- may discharge purulent-haematic material. ©SEEd Tutti i diritti riservati 96 Clinical Management Issues 2011; 5(3) S. Sabbatani, R. Manfredi, B. Fabbrizio, A. Caira, F. F. Trapani, G. Fasulo et al As examined by Urso and Farella in their association tenofovir-emtricitabine (200- 1996 contribution on Philoctetes’s disease 300 mg/day), plus the protease inhibitor [22], actually botryomycosis is primarily lo- atazanavir (300 mg/day), boostered with calized at limbs with cutaneous ulcers, has a ritonavir (100 mg/day), was recommended, long-term course in absence of an effective and taken by our patient with optimal adher- treatment, is complicated by purulent and ence and no relevant clinical and laboratory sero-hematic discharge, has an anamnestic adverse events. trauma, and a foul odour, but usually it is The past clinical history of our patient in- not painful. cluded a previous,