Unusual manifestation of disseminated herpes simplex virus type 2 infection associated with pharyngotonsilitis, esophagitis, and hemophagocytic lymphohisitocytosis without genital involvement View Full Text


Ontology type: schema:ScholarlyArticle      Open Access: True


Article Info

DATE

2019-01-17

AUTHORS

Shuhei Kurosawa, Noritaka Sekiya, Kazuaki Fukushima, Kazuhiko Ikeuchi, Akito Fukuda, Hideyuki Takahashi, Fangyi Chen, Hideki Hasegawa, Harutaka Katano, Tsunekazu Hishima, Keigo Setoguchi

ABSTRACT

BACKGROUND: Herpes simplex virus (HSV) has various presentations, depending on the patient's immune status, age, and the route of transmission. In adults, HSV type 1 is found predominantly in the oral area, and HSV type 2 (HSV-2) is commonly found in the genital area. HSV-2 infection without genital lesions is uncommon. Herein we report a unique case of pharyngotonsillitis as an initial manifestation of disseminated HSV-2 infection without genital involvement. CASE PRESENTATION: A 46-year-old male was admitted to our hospital with a 1-week history of fever and sore throat. His past medical history included hypereosinophilic syndrome diagnosed at age 45 years. Physical examination revealed throat congestion, bilaterally enlarged tonsils with exudates, tender cervical lymphadenopathy in the left posterior triangle, and mild epigastric tenderness. The laboratory data demonstrated bicytopenia, elevated liver enzyme levels, and hyperferritinemia. A bone marrow smear showed hypocellular marrow with histiocytes and hemophagocytosis. The diagnosis of HLH was confirmed, and the patient was treated with methylprednisolone pulse therapy on days 1-3. On day 5, despite initial improvement of the fever and sore throat, multiple, new, small bullae developed on the patient's face, trunk, and extremities. Additional testing showed that he was positive for HSV-specific immunoglobulin M and immunoglobulin G. Disseminated HSV infection was suspected, and intravenous acyclovir (10 mg/kg every 8 h) was begun. A subsequent direct antigen test of a bulla sample was positive for HSV-2. Moreover, tonsillar and esophageal biopsies revealed viral inclusion bodies. Immunohistochemical staining and a quantitative real-time polymerase chain reaction (PCR) assay confirmed the presence of HSV-2. Disseminated HSV-2 infection with multiple bullae, tonsillitis, esophagitis, and suspected hepatic involvement was diagnosed. After a 2-week course of intravenous acyclovir, his hematological status and liver function normalized, and his cutaneous skin lesions resolved. He was discharged on day 22 in good general health and continued taking oral valacyclovir for viral suppression due to his immunosuppressed status. CONCLUSION: Disseminated HSV-2 infection should be considered as one of the differential diagnoses in patients with pharyngotonsillitis and impaired liver function of unknown etiology even if there are no genital lesions. More... »

PAGES

65

Identifiers

URI

http://scigraph.springernature.com/pub.10.1186/s12879-019-3721-0

DOI

http://dx.doi.org/10.1186/s12879-019-3721-0

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1111492206

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/30654754


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20 schema:description BACKGROUND: Herpes simplex virus (HSV) has various presentations, depending on the patient's immune status, age, and the route of transmission. In adults, HSV type 1 is found predominantly in the oral area, and HSV type 2 (HSV-2) is commonly found in the genital area. HSV-2 infection without genital lesions is uncommon. Herein we report a unique case of pharyngotonsillitis as an initial manifestation of disseminated HSV-2 infection without genital involvement. CASE PRESENTATION: A 46-year-old male was admitted to our hospital with a 1-week history of fever and sore throat. His past medical history included hypereosinophilic syndrome diagnosed at age 45 years. Physical examination revealed throat congestion, bilaterally enlarged tonsils with exudates, tender cervical lymphadenopathy in the left posterior triangle, and mild epigastric tenderness. The laboratory data demonstrated bicytopenia, elevated liver enzyme levels, and hyperferritinemia. A bone marrow smear showed hypocellular marrow with histiocytes and hemophagocytosis. The diagnosis of HLH was confirmed, and the patient was treated with methylprednisolone pulse therapy on days 1-3. On day 5, despite initial improvement of the fever and sore throat, multiple, new, small bullae developed on the patient's face, trunk, and extremities. Additional testing showed that he was positive for HSV-specific immunoglobulin M and immunoglobulin G. Disseminated HSV infection was suspected, and intravenous acyclovir (10 mg/kg every 8 h) was begun. A subsequent direct antigen test of a bulla sample was positive for HSV-2. Moreover, tonsillar and esophageal biopsies revealed viral inclusion bodies. Immunohistochemical staining and a quantitative real-time polymerase chain reaction (PCR) assay confirmed the presence of HSV-2. Disseminated HSV-2 infection with multiple bullae, tonsillitis, esophagitis, and suspected hepatic involvement was diagnosed. After a 2-week course of intravenous acyclovir, his hematological status and liver function normalized, and his cutaneous skin lesions resolved. He was discharged on day 22 in good general health and continued taking oral valacyclovir for viral suppression due to his immunosuppressed status. CONCLUSION: Disseminated HSV-2 infection should be considered as one of the differential diagnoses in patients with pharyngotonsillitis and impaired liver function of unknown etiology even if there are no genital lesions.
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27 schema:keywords G. Disseminated HSV infection
28 HLH
29 HSV infection
30 HSV type 2
31 HSV-2
32 HSV-2 infection
33 HSV-specific immunoglobulin M
34 Herein
35 acyclovir
36 additional testing
37 adults
38 age
39 age 45
40 antigen test
41 area
42 bicytopenia
43 biopsy
44 body
45 bone marrow smears
46 bulla sample
47 bullae
48 cases
49 cervical lymphadenopathy
50 chain reaction
51 congestion
52 course
53 cutaneous skin lesions
54 data
55 day 1
56 day 22
57 day 5
58 diagnosis
59 diagnosis of HLH
60 differential diagnosis
61 direct antigen test
62 disseminated HSV infection
63 disseminated HSV-2 infection
64 disseminated herpes simplex virus type 2 infection
65 elevated liver enzyme levels
66 enzyme levels
67 epigastric tenderness
68 esophageal biopsies
69 esophagitis
70 etiology
71 examination
72 extremities
73 exudates
74 face
75 fever
76 function
77 general health
78 genital area
79 genital involvement
80 genital lesions
81 good general health
82 health
83 hematological status
84 hemophagocytic lymphohisitocytosis
85 hemophagocytosis
86 hepatic involvement
87 herpes simplex virus
88 herpes simplex virus type 2 infection
89 histiocytes
90 history
91 hospital
92 hypereosinophilic syndrome
93 hyperferritinemia
94 hypocellular marrow
95 immune status
96 immunoglobulin G. Disseminated HSV infection
97 immunoglobulin M
98 immunohistochemical staining
99 immunosuppressed status
100 impaired liver function
101 improvement
102 inclusion bodies
103 infection
104 initial improvement
105 initial manifestation
106 intravenous acyclovir
107 involvement
108 laboratory data
109 left posterior triangle
110 lesions
111 levels
112 liver enzyme levels
113 liver function
114 lymphadenopathy
115 lymphohisitocytosis
116 males
117 manifestations
118 marrow
119 marrow smears
120 medical history
121 methylprednisolone pulse therapy
122 mild epigastric tenderness
123 multiple bullae
124 oral area
125 oral valacyclovir
126 past medical history
127 patient's face
128 patient's immune status
129 patients
130 pharyngotonsilitis
131 pharyngotonsillitis
132 physical examination
133 polymerase chain reaction
134 posterior triangle
135 presence
136 presentation
137 pulse therapy
138 quantitative real-time polymerase chain reaction
139 reaction
140 real-time polymerase chain reaction
141 route
142 routes of transmission
143 samples
144 simplex virus
145 simplex virus type 2 infection
146 skin lesions
147 small bullae
148 smears
149 sore throat
150 staining
151 status
152 subsequent direct antigen test
153 suppression
154 syndrome
155 tender cervical lymphadenopathy
156 tenderness
157 test
158 testing
159 therapy
160 throat
161 throat congestion
162 tonsillitis
163 tonsils
164 transmission
165 triangle
166 trunk
167 type 1
168 type 2
169 type 2 infection
170 unique case
171 unknown etiology
172 unusual manifestation
173 valacyclovir
174 viral inclusion bodies
175 viral suppression
176 virus
177 virus type 2 infection
178 years
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