Full-text - Polski Przegląd Otorynolaryngologiczny

Full-text - Polski Przegląd Otorynolaryngologiczny

kazuistyka / case report
Tongue necrosis as a result of the overlap
syndrome – case report
Martwica języka w wyniku zespołu nakładania – opis
przypadku
Beata Piechnik1, Maciej Świetlicki1, Janusz Klatka1
1
Klinika Otolaryngologii i Onkologii Laryngologicznej Uniwersytetu Medycznego w Lublinie, kierownik: prof. dr hab. n. med. Janusz Klatka
Article history: Received: 12.11.2014 Accepted: 01.12.2014 Published: 28.02.2015
ABSTRACT:Tongue necrosis is very rare but very dramatic symptom, which occurs along with infection, injury, cancer or vasculitis.
We show the case of a patient with tongue necrosis as a result of the overlap syndrom of vasculitis and infection. Vasculitis can cause lower tissue perfusion and its hypooxia. There are many researches describing tongue necrosis as a rare
complication or first symptom of the Horton’s disease (giant cell arteritis of large and medium blood vessels). The beginning of the disease is rarely sudden, often insidiously, what makes all doctors to think about different causes of the
patient’s state. It was very similar situation in our case. Our patient was previously treated by many specialists: internist,
reumatologist and come to our ward in severe condition.
Treated with steroids patient got better very quickly. We must remember, however, about the possibility of many
causes, and we must try to treat those causes.
KEY WORDS: Horton’s disease, autoimmunization, infection
STRESZCZENIE: Martwica języka jest rzadkim, ale bardzo dramatycznym objawem chorobowym, który najczęściej pojawia się
w związku z infekcją, urazem, nowotworem złośliwym lub zapaleniem naczyń.
Przedstawiamy przypadek pacjentki, u której martwica języka mogła być skutkiem nakładania zapalenia naczyń i infekcji. Zapalenie naczyń krwionośnych powoduje obniżenie perfuzji tkanek, a co za tym idzie, ich niedotlenienie.
Wiele prac mówi o martwicy języka jako o rzadkim powikłaniu bądź też pierwszym objawie choroby Hortona, czyli
olbrzymiokomórkowego zapalenia naczyń krwionośnych dużego i średniego kalibru. Początek choroby rzadko bywa
nagły, zwykle rozwija się podstępnie, co ukierunkowuje myślenie lekarzy w inną stronę. Podobnie było w omawianym
przypadku. Pacjentka od dłuższego czasu była leczona internistycznie. Do szpitala trafiła w stanie ciężkim z powodu
infekcji bakteryjnej potwierdzonej badaniem bakteriologicznym podczas przyjmowania do szpitala.
Włączając sterydoterapię, zwykle uzyskuje się szybką poprawę stanu zdrowia pacjenta. Należy jednak pamiętać
o możliwości nakładania wielu przyczyn i w związku z tym należy się starać wdrożyć leczenie przyczynowe.
SŁOWA KLUCZOWE: choroba Hortona, autoimmunizacja, infekcja
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DOI: 10.5604/20845308.1136067
WWW.OTORHINOLARYNGOLOGYPL.COM
kazuistyka / case report
INTRODUCTION
Necrosis of the tongue is a rare yet dramatic pathological
symptom most commonly developing as a result of infection,
trauma, malignancy, or vasculitis [1,2,4,5,6,7]. Presented below is a case of a female patients in whom the probable causes of tongue necrosis included infection and vasculitis. It
is unknown whether both factors were independent of each
other or whether vasculitis was a result of infection (or just
an exacerbation of an ongoing autoimmune disease secondarily to the infection).
Fig. 1: Tongue necrosis.
CASE REPORT
A female patient, aged 53, was admitted in emergency to the
Clinic of Otolaryngology and Laryngological Oncology of the
Medical University due to suspected sepsis, inflammatory infiltration of the mouth floor and left parapharyngeal space.
Due to dyspnea, patient was subjected to tracheotomy in a local laryngology department. Upon admission, the patient was
in severe overall condition. Clinical examination revealed lockjaw, swelling of tongue and mouth floor, hard inflammatory
infiltration of submandibular and genial regions as well as the
left lateral surface of the neck, extending down towards the
chest. Medical history included treatment of rheumatoid arthritis, recurrent lower limb thrombosis, normocytic anemia
with leukopenia, arterial hypertension, thoracic osteoarthritis.
Wide-lumen drainage of the inflammatory infiltration in the
left parapharyngeal space was performed on the day of admission. Massive serous inflammation was detected within
soft tissues. Additional investigations revealed high levels of
procalcitonin and inflammatory markers (CRP, leukocytosis).
Neck drains were removed on the fourth day of hospitalization at the Clinic. During her stay in the clinic, the patient
was subject to multiple consultations by a rheumatologist,
internal medicine specialist, anesthesiologist, thoracic surgeon, angiologist and hematologist. Following the surgical
and conservative treatment (including antibiotic and steroid therapy), patient’s overall and local condition gradually
improved. On the fifth day of hospitalization, spontaneous
rupture of the tongue body occurred on the left. A focal necrotic lesion sized ca. 2x1.5 cm was revealed upon examination and removed from within the wound in the tongue
body. Cleansing the tongue and mouth of necrotic tissue was
continued over the several following days. During the period,
the patient was fed via a gastric tube. Due to the low values
of erythrocytic parameters, the patient received a transfer
of 2 units of red blood concentrate of compatible type, absorbed without complications. On the 15th day of hospitaPOLSKI PRZEGLĄD OTORYNOLARYNGOLOGICZNY, TOM 4, NR 1 (2015), s. 36-39
lization, the patient was decannulated and the tracheotomy
opening was closed surgically.
The patient was discharged home in satisfactory general condition, respiratorily and circulatorily efficient, in full logical
contact, under family care with recommendations of continued treatment in internal medicine, rheumatology, hematology and laryngology outpatient clinics.
DISCUSSION
Causes of tongue necrosis may vary. Besides those listed
below, literature reports cases of extended intubation (necrosis as a reaction to the presence of intubation drain) [2].
Vasculitis of various etiology, with all possible consequences, is
considered to be the most common cause of tongue necrosis.
The patient presented above had been undergoing treatment
for rheumatoid disease, and thus vasculitis was proposed as the
suspected cause of tongue necrosis. Vasculitis leads to reduced
tissue perfusion and thus hypoxia. Numerous studies report
tongue necrosis to be a rare complication or the first symptom
of Horton disease, i.e. giant-cell arteritis of large- and middlesized arteries [4,5,6,7,8]. The disease usually affects individuals
above the age of 50. The incidence in female is observed to be
higher than in males, usually at a ratio of ca. 2:1 [9]. The probable cause of Horton disease consists in an immune reaction
to infectious agents in genetically predisposed individuals [10].
No serological markers of autoimmunization are observed. Similar clinical presentation was observed in the reported case
where inflammation (probably due to infection) and elevated
laboratory parameters (procalcitonin, CRP, leukocytosis) was
observed. The result of serological screening for the markers
of autoimmunization was negative. The onset of the disease
may be rapid, with the disease usually developing insidiously
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kazuistyka / case report
and directing the attention of the physicians to other possible
causes. The reported case was similar. The patient had been
treated at an internal medicine clinic for quite a long time. She
was admitted to hospital in severe condition due to bacterial
infection as confirmed by bacteriological screening upon admission (Streptococcus pyogenes in the smear collected from
tongue wound after removal of necrotic tissue). The infection
might also have been the cause triggering the rapid acceleration
of the natural history of the underlying disease. Diagnosing the
giant cell vasculitis may be challenging for the attending team.
Also the histopathological presentation of arterial biopsy may
be non-diagnostic [11]. In case of Horton disease, the literature reports other, more common symptoms suggestive of this
disorder, including ocular symptoms (including blindness),
features of peripheral arthropathy, new-onset headaches, or
swollen extremities [12]. These symptoms were not observed
in the reported case, although the medical history included
the treatment for rheumatoid arthritis.
Tongue necrosis may also be due to acute cardiac insufficienncy leading to acute hypoxia of multiple organs, including head and neck organs. Usually, such a case is associated with unfavorable prognosis [3]. This suspicion was one
of the cause of multiple specialist consultations of the case
carried out during the patient’s stay at the Clinic.
SUMMARY
Tongue necrosis is always a severe condition posing significant diagnostic difficulties. Acute organ ischemia, myocardial infarction, and Horton disease should be considered
as possible causes of the symptom. Introduction of steroid
therapy usually leads to fast improvement in patient’s condition. However, one should keep in mind the possibility of
overlapping of multiple causes and thus attempt to initiate
causal treatment.
References
1.
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2.
Kuhn MA, Zeitler DM, Myssiorek DJ Tongue necrosis: a rare complication of oral intubation.. Laryngoscope. 2010;120 Suppl 4:S159. doi: 10.1002/lary.21623.
3.
Roman BR, Immerman SB, Morris LG. Ischemic necrosis of the tongue in patients with cardiogenic shock. Laryngoscope. 2010 Jul;120(7):1345-9. doi:
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Papadopoulos E, Szurman P, Haberl GC, Tudor SE, Petermeier K. Tongue necrosis as first clinical manifestation of Horton giant cell arteritis .Klin Monbl
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Husein-Elahmed H, Callejas-Rubio JL, Rios-Fernández R, Ortego-Centeno N. Tongue infarction as first symptom of temporal arteritis. Rheumatol Int. 2012
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doi: 10.1007/s10067-009-1141-z. Epub 2009 Mar 10.
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Sainuddin S, Saeed NR. Acute bilateral tongue necrosis--a case report. Br J Oral Maxillofac Surg. 2008 Dec;46(8):671-2. doi: 10.1016/j.bjoms.2008.03.027.
Epub 2008 May 21.
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Schurr C, Berthele A, Burghartz M, Kiefer J. Spontaneous bilateral necrosis of the tongue: a manifestation of giant cell arteritis? Eur Arch Otorhinolaryngol.
2008 Aug;265(8):993-8. doi: 10.1007/s00405-007-0556-x. Epub 2008 Jan 23.
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Emmerich J., Fiessinger JN.. Epidemiology and etiological factors in giant cell arteritis (Horton’s disease and Takayasu’s disease).. „Ann Med Interne (Paris)”.
149 (7), s. 425-32, Nov 1998.
10. Rozwodowska M., Rozwodowska MM., El-Essa A., Kubica J. Olbrzymiokomórkowe zapalenie tętnic (zapalenie tętnicy skroniowej). Opis 2 przypadków.
„Choroby Serca i Naczyń”. nr 4, s. 211–216, 2006.
11. Pfeiffer J, Ridder GJ. Spontaneous tongue necrosis consecutive to rheumatoid hyperviscosity syndrome. A case report and literature review. Laryngorhinootologie. 2008 Jan;87(1):43-8. Epub 2007 Aug 23.
12. Salvarani C, Hunder GG. Musculoskeletal manifestations in a population-based cohort of patients with giant cell arteritis. „Arthritis Rheum.”. 42 (6), s. 1259–
66, June 1999.
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kazuistyka / case report
Word count: 909 Tables: – Figures: 1 References: 12
Access the article online: DOI: 10.5604/20845308.1136067 Full-text PDF: www.otorhinolaryngologypl.com/fulltxt.php?ICID=1136067
Corresponding author: Beata Piechnik, Klinika Otolaryngologii i Onkologii Laryngologicznej Uniwersytetu Medycznego w Lublinie,
ul. Jaczewskiego 8, 20-954 Lublin, tel. 81 7244518, e-mail: [email protected], tel. kom. 502566936
Copyright © 2015 Polish Society of Otorhinolaryngologists Head and Neck Surgeons. Published by Index Copernicus Sp. z o.o. All rights reserved Funding: No additional funding was provided during creation of the manuscript.
Competing interests: The authors declare that they have no competing interests.
Cite this article as: Piechnik B., Świetlicki M., Klatka J.: Tongue necrosis as a result of the overlap syndrome – case report. Pol Otorhino Rev 2015; 4(1): 36-39
POLSKI PRZEGLĄD OTORYNOLARYNGOLOGICZNY, TOM 4, NR 1 (2015), s. 36-39
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