Sporotrichosis in pregnancy: case reports of 5 patients in a zoonotic epidemic in Rio de Janeiro, Brazil

Abstracts

Os autores apresentam cinco casos de esporotricose em gestantes numa epidemia zoonótica no Rio de Janeiro. São discutidos principalmente os aspectos clínicos e as dificuldades na escolha terapêutica desse grupo específico de pacientes.

Anfotericina B; Esporotricose; Gestantes; Resultado de tratamento; Terapêutica

CASE REPORT

Sporotrichosis in pregnancy: case reports of 5 patients in a zoonotic epidemic in Rio de Janeiro, Brazil^*

Rosane Orofino Costa^I; Andrea Reis Bernardes-Engemann^II; Luna Azulay-Abulafia^III; Fabiana Benvenuto^IV; Maria de Lourdes Palermo Neves^V; Leila Maria Lopes-Bezerra^VI

^IPhD - Adjunct Professor at the State University of Rio de Janeiro (Universidade do Estado do Rio de Janeiro - Rio de Janeiro (RJ), Brazil

^IIPhD - Biologist at the State University of Rio de Janeiro (Universidade do Estado do Rio de Janeiro - Rio de Janeiro (RJ), Brazil

^IIIPhD - Adjunct Professor at the State University of Rio de Janeiro (Universidade do Estado do Rio de Janeiro - Rio de Janeiro (RJ), Brazil

^IVBiologist - Federal University of the State of Rio de Janeiro (Universidade Federal do Estado do Rio de Janeiro - UERJ) - Rio de Janeiro (RJ), Brazil

^VMaster's degree student at the State University of Rio de Janeiro ( Universidade do Estado do Rio de Janeiro - Rio de Janeiro (RJ), Brazil

^VIPhD student - Adjunct Professor at the State University of Rio de Janeiro (Universidade do Estado do Rio de Janeiro - Rio de Janeiro (RJ), Brazil

^{Mailing address}

ABSTRACT

Five cases of sporotrichosis occurring in pregnant women in a zoonotic epidemic in Rio de Janeiro, Brazil, are described. The main clinical features, as well as the challenging therapeutic choices for this specific group of patients, are discussed.

Keywords: Amphotericin B; Pregnant women; Sporotrichosis; Therapeutics; Treatment outcome

INTRODUCTION

Sporotrichosis has always been regarded as an occupational disease with a strong rural profile. Nevertheless, since 1997 the city of Rio de Janeiro has been experiencing an epidemic of zoonotic transmission by felines, considered rare in other parts of the world and that had a previous rate of circa one case/year. Marques and cols.¹ published a report, in 1998, about two feline transmission cases observed in a couple living in a rural area of the interior of the state of São Paulo. A wide diversity of clinical presentations has been observed, including the extracutaneous forms, uncommon before the outbreak of this epidemic in 1997. Some risk groups, such as elderly patients, those infected with AIDS or other comorbidities evolve with severe clinical manifestations, oftentimes fatal. Women, who usually take care of domestic animals, are more susceptible to the infection. We report the occurrence of five clinical cases of this mycosis in pregnant women, analyzing its clinical and serological aspects, as well as the difficulty faced when making a therapy choice.

CASE REPORTS

During the period from 1997 to 2009, 171 cases of sporotrichosis were diagnosed in the University Hospital Pedro Ernesto, RJ, five of them in pregnant women. Four of these patients reported contact with a sick animal (cat) and one of them with an apparently healthy cat. One of the patients was a veterinarian and contracted the disease when professionally caring for the animal. She began the treatment by taking terbinafine and found out she was pregnant while using oral terbinafine. Delivery was normal after a fullterm pregnancy. Gestational age varied from 3 to 24 weeks and the mean age of these patients was 29.2 years, with a predominance of 80% of the lymphocutaneous form; none of them had any sign of systemic disease, except for polyarthritis diagnosed as reactive arthritis (Figure 1). The diagnoses of all cases were confirmed by isolation of S. schenckii from the cutaneous lesion. Serological tests were also carried out according to methodology described by Bernardes-Engemann and col. that consists in detection of IgG antibodies against the antigen SsCBF, isolated from the S. Schenckii cell wall.² The highest titers were found in the patient with more extensive clinical manifestations; in the others the titers were lower. There was one false-negative titer. Three patients were treated with systemic antifungal medication and two only with thermotherapy, a conservative treatment (Figures 2 and 3). All of the patients had full term pregnancies and, except for one who did not receive systemic treatment, their children were born healthy. The death of one child had no relationship with sporotrichosis. There was no need to continue the treatment for any of the patients after delivery, as all were cured. The most important aspects of the clinical cases are found in Chart 1.

DISCUSSION

The clinical symptoms of pregnant women with human sporotrichosis transmitted by cat do not differ from the classical ones, with predominance of lymphocutaneous forms. In the series of cases described by Barros and col., of the 178 patients with sporotrichosis examined between 1998-2001 only one patient was pregnant (0.56%) and she was treated with thermotherapy, with total resolution of the lesions presented.³ In our case study, there was an increase in the number of pregnant women infected; the studied period was somewhat longer. Of 171 patients diagnosed between 1997-2009, five were pregnant (2.92%). Nevertheless, it can still be considered an infectious disease uncommon during pregnancy. Plauché described a pregnant woman treated with potassium iodide in 1986; despite the contraindications the child was born at term and healthy.⁴ Roming and col. in 1972, as well as Vanderveen and col. in 1982 described thermotherapy as a therapeutic option to treat sporotrichosis during pregnancy.^5,6 More recently, in a report of nine sporotrichosis cases published by Agarwal and col. in 2008, one of the patients was pregnant (11.11%) and was treated with local heat, after which the lesions were healed.⁷ Local heat therapy is based on fungi thermotolerance and was first described for treatment of chromomycosis by Tagami and col., in 1979.⁸ At present, it may be indicated as an effective therapeutic option in subcutaneous mycoses in an otherwise healthy individual, with localized lesions. The treatment of choice should be based on clinical presentation, the immune status of the pregnant woman and the possibility of risks for mother and fetus, both from the medication and from the infection.⁹ During pregnancy, the ideal is not to prescribe medication, mainly if the disease is not going to affect the health of mother and child. Therefore, it is important for the specialists involved obstetrician, dermatologist, infectologist - to get together so that there is ample discussion and consensus about the need to establish a treatment, especially if systemic; this was our conduct. Azolic agent therapy in pregnancy should be avoided, due to its teratogenic and embryotoxic potential, since all are considered as category C; the use of saturated potassium iodide solution is contraindicated, since it is associated with neonatal hypothyroidism, thyromegaly, fetal respiratory obstruction and prolonged delivery, being counted among category D drugs. There are no reports on the use of terbinafine in pregnancy, as it is considered category B, which means that there are no studies regarding its safety in this group of patients and therefore its effects on the fetus are unknown.¹⁰ One patient in our study who was treated with terbinafine became pregnant in the course of the sporotrichosis treatment, that is, its use during pregnancy was not intentional. Amphotericin B is the most effective drug for the treatment of systemic sporotrichosis. There are no reports on harmful effects to the fetus and it is recommended in the treatment of severe sporotrichosis during pregnancy, although the frequency of its adverse effects, such as hyperpyrexia, nausea, vomiting, anemia and renal toxicity limits its choice in the treatment of localized disease. In the series of cases here reported there was no maternalfetal damage with any of the therapeutic approaches. The death of an infant does not seem to be related to the infection, as the lesion was regressing spontaneously and the pregnant woman did not receive systemic medication but only local heat. In this case, drug therapy could have been implicated as the cause of fetal death. It is still not clear whether there is any fungal interference effect on the fetus, and a possible placental transfer is unknown. It is not possible to foresee, in a pregnant woman, the consequences of a more severe extracutaneous infection and its treatment, mainly such as have been seen in immunosuppressed patients during the epidemic. Currently, only two forms of treatment may be recommended to pregnant women: local heat or amphotericin B.¹¹ If the lesion is small and localized, and the pregnant woman does not present other intercurrences, the ideal strategy is to adopt a conservative treatment with local heat until the time of delivery, when the clinical progression can be reevaluated. The serological tests carried out by our group have shown good clinical and evolutive correlation. They are especially useful in cases of difficult diagnosis as a screening tool, or for follow-up of uncommon cases, as serum and other organic fluids, such as synovial liquid, may be used.^2,12 High titers have been shown in cases of disseminated disease. This case report has the objective of exposing therapeutic conduct difficulties for an emerging disease in special situations and calling attention to a new risk group in this epidemic.

REFERENCES

1. Marques SA, Camargo RMP, Haddad Junior V, Marques MEA, Rocha NS, Franco SRVS. Esporotricose humana: transmitida por felino. An Bras Dermatol. 1998;73:559-64.

2. Bernardes-Engemann AR, Costa RC, Miguens BR, Penha CV, Neves E, Pereira BA, et al. Development of an enzyme-linked immunosorbent assay for the serodiagnosis of several clinical forms of sporotrichosis. Med Mycol. 2005;43:487-93.

3. Barros MB, Schubach Ade O, do Valle AC, Gutierrez Galhardo MC, Conceição-Silva F, Schubach TM, et al. Cat-transmitted sporotrichosis epidemic in Rio de Janeiro, Brazil: description of a series of cases. Clin Infect Dis. 2004;38:529-35.

4. Plauché WC. Sporotrichosis in pregnancy. Am J Obstet Gynecol. 1968;100:1150-1.

5. Romig DA, Voth DH, Liu C. Facial sporotrichosis during pregnancy. A therapeutic dilemma. Arch Intern Med. 1972;130:910-2.

6. Vanderveen EE, Messenger AL, Voorhees JJ. Sporotrichosis in pregnancy. Cutis. 1982;30:761-3.

7. Agarwal S, Gopal K, Umesh, Kumar B. Sporotrichosis in Uttarakhand (India): a report of nine cases. Int J Dermatol. 2008;47:367-71.

8.Tagami H, Ohi M, Aoshima T, Moriguchi M, Suzuki N, Yamada M. Topical heat therapy for cutaneous chromomycosis. Arch Dermatol. 1979;115:740-1.

9. King CT, Rogers PD, Cleary JD, Chapman SW. Antifungal therapy during pregnancy. Clin Infect Dis. 1998;27:1151-60.

10. Bennett JE. Antifungal Agents. In: Brunton, LL, Lazo JS, Parker KL, editors. Goodman and Gilman′s The Pharmacological Basis of Therapeutics. 11th ed. New York: McGraw-Hill; 2006. p.1240.

11. Kauffman CA, Bustamante B, Chapman SW, Pappas PG; Infectious Diseases Society of America. Clinical practice guidelines for the management of sporotrichosis: 2007 update by the Infectious Diseases Society of America. Clin Infect Dis. 2007;45:1255-65.

12. Costa RO, de Mesquita KC, Damasco PS, Bernardes-Engemann AR, Dias CM, Silva IC, et al. Infectious arthritis as the single manifestation of sporotrichosis: serology from serum sample and synovial fluid as an aid to diagnosis. Rev Iberoam Micol. 2008;25:54-6.

Received on 03.03.2010.

Approved by the Advisory Board and accepted for publication on 21.06.2010.

Conflict of interest: None

Financial funding: Ministry of Health/Faperj

*

Studied carried out at the Dermatology Service - University Hospital Pedro Ernesto - State University of the State of Rio de Janeiro (HUPE - UERJ) and Laboratory of Cell Mycology and Proteomics at the Department of Cellular Biology and Genetics of the Institute of Biology Roberto Alcântara Gomes (IBRAG - UERJ) - Rio de Janeiro (RJ), Brazil.

Publication Dates

• Publication in this collection
  01 Dec 2011
• Date of issue
  Oct 2011

History

• Received
  03 Mar 2010
• Accepted
  21 June 2010