Case #16008

A young man with a diffuse rash after travel to Madagascar.

History of Present Illness

2016 IDWeek Fellows' Day Case Presentation: A man in his twenties developed a diffuse rash shortly after a two-week trip to Madagascar.

One week before the onset of the rash, when he was on a beach on the southern coast in Madagascar, he developed a frontal headache (rated 4 out of 10, on a scale of 0 to 10 where 10 is the most severe) that increased with movement. At that time, he also noticed a lesion on his left upper arm that appeared to be an insect bite.  Two days later, chills and sore throat occurred, and the skin lesion developed a scab and became progressively darker.  Six days after the onset of the headache, a rash developed on his arms, chest, legs, and face. 

The next day, he took ibuprofen for his symptoms, and went to the hospital where he was admitted. He reported no cough, photophobia, abdominal pain, sweating or dysuria.

Past Medical History

The patient reported having had chickenpox when he was child.

Medications

He took ibuprofen (600 mg orally). He had not taken malaria prophylaxis while traveling.

Epidemiological History

He lived in the Northeastern United States and worked in an office. He was sexually active, without use of barrier protection, with two female partners in the previous six months. He drank alcohol socially and did not smoke cigarettes or use illicit drugs. His sibling and friends did not have symptoms. 

Physical Examination

He was well nourished and not in distress. The blood pressure was 120/80 mm Hg, pulse 72 beats per minute, temperature 98.6°F (37°C), and respirations 16 per minute. There were scattered vesiculopapular lesions over the trunk, upper and lower extremities, which spared the palms and soles. The lesions were surrounded by erythema that blanched to palpation. There was an eschar (6 mm in diameter) on his left arm that was non-tender and had an erythematous rim (Figure 1).  A  mobile non-tender lymph node (1 cm diameter) was palpable in the left axilla. The remainder of the examination was normal.

Studies

The white cell count was 5900 per cubic millimeter (reference range 150,000-450,000), with 48% neutrophils, 33.8% lymphocytes, 17.2% monocytes, and 0.4% eosinophils. The blood level of total bilirubin was 1.6 mg/dL (reference range 0.3-1.2) and direct bilirubin 0.2 mg/dL (reference range 0.05-0.1).  The hematocrit, platelet count, and blood levels of electrolytes, creatinine, alkaline phosphatase, and transaminases were normal.  There were no intraerythrocytic or extra-erythrocytic parasites seen on peripheral smear.  Cultures of the blood and urine were obtained.

A chest radiograph (Figure 2) was normal.

What is the diagnosis?

 

Diagnostic Procedure(s) and Result(s)

The eschar lesion on the left arm was biopsied, and pathological examination of the specimen with hematoxylin and eosin (H and E) staining showed dense perivascular lymphohistiocytic infiltrates (Figures 3 through 5).  Figure 3 (low power field of the skin biopsy) revealed a superficial and deep angiocentric adnexotropic lymphohistiocytic infiltrate with attendant ulceration.  Figure 4 (higher power magnification) showed the dominant mononuclear cell nature of the infiltrate and its apposition to vessels and nerves. Figure 5 revealed frank vasculitic changes characterized by an angiocentric lymphohistiocytic infiltrate surrounding and permeating vessels with evidence of vascular injury characterized by fibrin deposition and red cell extravasation. Figure 6 demonstrated concomitant interface dermatitis with lymphocyte tagging along the dermal-epidermal junction and serpentine-like macrophages. These findings were thought to be suggestive of Rickettsialpox.

The skin biopsy specimen was sent to the Center for Disease Control and Prevention where the molecular and immunohistochemical testing confirmed the presence of Rickettsia akari.

Serologic testing for Rocky Mountain spotted fever revealed negative IgG (<1:64) but positive IgM (>1:64). Testing for HIV nucleic acid was undetectable (<20 copies/mL). Rapid plasma reagin (RPR) was non-reactive. Varicella zoster serology was consistent with remote immunity with negative IgM and positive IgG. Two sets of blood cultures and a urine culture were negative.

Treatment and Followup

Doxycycline (100 mg twice daily, orally) and valacyclovir (1 g every 8 hours) were administered pending test results.  Physical findings after two days of therapy (Figure 7) are shown; the erythema surrounding the left arm eschar had improved.

Valacyclovir was stopped after the varicella zoster virus direct fluorescent antibody (DFA) test came back negative. Varicella zoster virus culture was also negative.

The patient completed a 1-week course of doxycycline twice daily.  Follow-up 1 month later revealed complete resolution of the skin rash with a hypo-pigmented scar at the eschar site.

Discussion

Rickettsialpox is caused by Rickettsia akari, which is transmitted by the bite of the house-mouse mite Liponyssoides sanguineus [Huebner, Stamps and Armstrong, 1946; Huebner et al., 1947; Huebner, Jellison and Pomerantz., 1946]. The disease was named rickettsialpox because of its resemblance to chickenpox [Huebner, Stampls and Armstrong, 1946]. It occurs primarily in urban areas, where the density of mites, mice, and humans is high [Koss et al., 2003]. Huebner and his collaborators first isolated Rickettsia akari from a patient, mites, and a naturally infected house mouse in Queens, New York in 1946 [Huebner, Stamps and Armstrong, 1946; Huebner et al., 1947; Huebner, Jellison and Pomerantz., 1946].

Rickettsialpox is almost certainly under-recognized and under-reported [Koss et al., 2003]. The bioterrorism attacks of October 2001 may have led to increased awareness and detection of this disease [Koss et al., 2003]. Prior to that time, these clinical syndromes may have been misdiagnosed, or perhaps the infected persons did not seek medical attention. Since the 1960s, only single case reports and small case series have been published, and most of these cases have occurred in New York City [Huebner, Stamps and Armstrong, 1946; Huebner et al., 1947; Huebner, Jellison and Pomerantz., 1946; Koss et al., 2003; Kass et al., 1994], but the disease has also been reported in other large cities in the eastern United States [Koss et al., 2003; Kass et al., 1994] as well as in Europe, South Africa and Latin America [Parola et al., 2013]. The presence of an eschar is a key clinical feature of rickettsialpox [Huebner, Stamps and Armstrong, 1946; Huebner et al., 1947]. This develops at the location of a mite bite, followed by the onset of systemic symptoms and a more generalized papulovesicular rash [Huebner, Stamps and Armstrong, 1946; Huebner et al., 1947; Huebner, Jellison and Pomerantz., 1946].

The most common disease that mimics rickettsialpox is chickenpox [Huebner, Stamps and Armstrong, 1946]. However, rickettsialpox characteristically has an eschar whereas chickenpox does not. In addition, in rickettsialpox, the rash consists of papulovesicles whereas in chickenpox the rash is vesicular and appears in crops [Heininger et al., 2006]. Finally, chickenpox usually presents with many more skin lesions than rickettsialpox.

Rickettsialpox has also been confused with cutaneous anthrax. This underscores the importance of clinicians' ability to promptly recognize and definitively diagnose rickettsialpox [Koss et al., 2003]. Patients with cutaneous anthrax characteristically have local edema out of proportion to the size of the lesions and gram stain of these lesions should reveal nonsporulating gram-positive rods [CDC 2001]. Rickettsialpox can also be confused with other rickettsial diseases, including Mediterranean spotted fever caused by Rickettsia conorii [Raoult et al., 1986], African tick bite fever [Owen et al., 2006], Rickettsia parkeri infection [Parola et al., 2013], and scrub typhus [Paddock et al., 2006]. All of these infections may be associated with eschars, but only African tick bite fever has rash that is typically or occasionally vesicular [Owen et al., 2006; Parola et al., 2013]. Rickettsia africae, the agent of African tick bite fever, is endemic in Africa - including Madagascar where our patient had recently travelled [Parola et al., 2013] - which made the diagnosis of our patient challenging.

The diagnosis of rickettsialpox is mainly a clinical one based on characteristic symptoms and signs [Huebner, Stamps and Armstrong, 1946; Huebner et al., 1947; Huebner, Jellison and Pomerantz., 1946; Koss et al., 2003]. It can be established retrospectively by documenting a fourfold rise in convalescent titers of complement fixation or indirect fluorescent antibodies using spotted fever group antigens [Koss et al., 2003]. Direct immunofluorescence can be used to identify rickettsial organisms in paraffin-embedded biopsy tissue from patients with rickettsialpox [Kass et al., 1994]. A definitive diagnosis can be made by isolation of Rickettsia akari from skin eschars [Paddock et al., 2006], or by a real-time multiplex polymerase chain reaction (PCR) that can specifically identify Rickettsia akari in formalin-fixed paraffin-embedded skin biopsy specimens [Denison et al., 2014], as in our case. Such tests are available through most state health departments and the Centers for Disease Control and Prevention [Koss et al., 2003].

Most patients with rickettsialpox have a mild illness and full recovery can be expected even without treatment [Kass et al., 1994]. The treatment of choice for rickettsialpox is doxycycline (100 mg orally every 12 hours) until the patient has been afebrile and clinically well for 48 to 72 hours [Kass et al., 1994].

In conclusion, rickettsialpox should be considered as a differential diagnosis in the setting of an eschar and a papulovesicular rash. Clinicians should be familiar with the clinical presentation and diagnostic features of rickettsialpox because it may be confused with more serious diseases, such as cutaneous anthrax [Koss et al., 2003] or chickenpox [Huebner, Stamps and Armstrong, 1946]. It is a self-limited disease; however, antibiotics hasten defervescence and provide relief of other systemic symptoms.

Final Diagnosis

Rickettsialpox caused by Rickettsia akari.

References
  1. Centers for Disease Control and Prevention (CDC). (2001) Update: Investigation of bioterrorism-related anthrax and interim guidelines for clinical evaluation of persons with possible anthrax. MMWR Morb Mortal Wkly Rep. 2001;50(43):941. PMID:11708591 (PubMed abstract)
  2. Denison AM, Amin BD, Nicholson WL, Paddock CD. (2014) Detection of Rickettsia rickettsii, Rickettsia parkeri, and Rickettsia akari in skin biopsy specimens using a multiplex real-time polymerase chain reaction assay. Clin Infect Dis. 2014 Sep;59(5):635-42. PMID:24829214 (PubMed abstract)
  3. Heininger U, Seward JF. (2006) Varicella. Lancet. 2006;368(9544):1365-1376. PMID:17046469 (PubMed abstract)
  4. Huebner RJ, Stamps P and Armstrong C. (1946) Huebner RJ; Jellison WL and Armstrong C. (1946) Rickettsialpox; a newly recognized rickettsial disease; isolation of the etiological agent. Public Health Rep. 1946 Nov 8;61(45):1605-14. PMID:20341090 (PubMed abstract)
  5. Huebner RJ; Jellison WL and Armstrong C. (1947) Rickettsialpox; a newly recognized rickettsial disease; recovery of Rickettsia akari from a house mouse (Mus musculus). Public Health Rep. 1947 May 30;62(22):777-80. PMID:20342271 (PubMed abstract)
  6. Huebner RJ; Jellison WL and Pomerantz C. (1946) Rickettsialpox, a newly recognized rickettsial disease; isolation of a Rickettsia apparently identical with the causative agent of rickettsialpox from Allodermanyssus sanguineus, a rodent mite. Public Health Rep. 1946 Nov 22;61(47):1677-82. PMID:20341097 (PubMed abstract)
  7. Kass EM, Szaniawski WK, Levy H, Leach J, Srinivasan K, Rives C. (1994) Rickettsialpox in a New York City hospital, 1980 to 1989. N Engl J Med. 1994;331(24):1612-1617.. PMID:7969341 (PubMed abstract)
  8. Koss T, Carter EL, Grossman ME, Silvers DN, Rabinowitz AD, Singleton J Jr, Zaki SR, Paddock CD. (2003) Increased detection of rickettsialpox in a New York City hospital following the anthrax outbreak of 2001: use of immunohistochemistry for the rapid confirmation of cases in an era of bioterrorism. Arch Dermatol. 2003 Dec;139(12):1545-52. PMID:14676069 (PubMed abstract)
  9. Owen CE, Bahrami S, Malone JC, Callen JP, Kulp-Shorten CL. (2006) African tick bite fever: a not-so-uncommon illness in international travelers. Arch Dermatol. 2006;142(10):1312. PMID:17043186 (PubMed abstract)
  10. Paddock CD, Koss T, Eremeeva ME, Dasch GA, Zaki SR, Sumner JW. (2006) Isolation of Rickettsia akari from eschars of patients with rickettsialpox. Am J Trop Med Hyg. 2006 Oct;75(4):732-8. PMID:17038703 (PubMed abstract)
  11. Parola P, Paddock CD, Socolovschi C, Labruna MB, Mediannikov O, Kernif T, Abdad MY, Stenos J, Bitam I, Fournier PE, Raoult D. (2013) Update on tick-borne rickettsioses around the world: a geographic approach. Clin Microbiol Rev. 2013 Oct;26(4):657-702. PMID:24092850 (PubMed abstract)
  12. Raoult D, Weiller PJ, Chagnon A, Chaudet H, Gallais H, Casanova P. (1986) Mediterranean spotted fever: clinical, laboratory and epidemiological features of 199 cases. Am J Trop Med Hyg. 1986;35(4):845-850. PMID:3728799 (PubMed abstract)
Notes

This case was contributed by Maroun Sfeir MD MPH, Cynthia Magro MD and Sian Jones MD of the New York Presbyterian Weill Cornell Medical Center, New York, NY.

The case was originally presented at IDWeek 2016, a joint effort of Infectious Diseases Society of America (IDSA), HIV Medical Association (HIVMA), Pediatric Infectious Diseases Society (PIDS), and the Society for Healthcare Epidemiology of America (SHEA), during an interactive session on Fellows' Day.

Figure 1, 2, and 7 were provided by Dr. Sfeir and Figures 3, 4, 5 and 6 were provided by Dr. Magro.

Copyright Infectious Disease Society of America (IDSA), 2016. Used with permission.

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