The Journal of Craniofacial Surgery  Volume 28, Number 3, May 2017
infections may occur more often after the celebration of the feast of
the sacrifice in Muslim countries.
Key Words: Nose, orf, parapoxvirus
O rf is a zoonotic viral disease caused by orf virus. Orf virus is a
member of the genus parapoxvirus in the family Poxviridae.
Generally, the orf virus is transmissible to humans via direct contact
with infected animals. Infection manifests as localized skin lesions.
Most cutaneous lesions are self-limited and resolve within 6 to 10
weeks.1 The most common site of infection is the hand. However, the
nose has been described as a rare location of infection.2 We present a
rare case of a patient with orf infection presenting as a nasal tip mass.
CLINICAL REPORT
A 52-year-old Turkish man presented to our clinic with a 25-day
history of mass on his nose. He had been diagnosed with infective
dermatitis and empirically treated with oral cefdinir and topical
mupirocin for 6 days. Afterward, he was referred to our hospital
because his symptoms still persisted and the mass size increased
despite treatment. His medical history was nonspecific. More specifi-
cally, he had no history of nasal trauma, insect bites, or nasal surgery.
The clinical examination revealed a painless 2  1.7 cm
immobile mass with erythema at the tip of his nose (Fig. 1A).
The bilateral anterior rhinoscopy was normal. The dermatological
examination revealed a 1  1 cm diameter papule on the index
finger extensor surface of right hand (Fig. 1B).
The patient indicated that he had contact with sheep using
ungloved hands on the first day of the feast of the sacrifice, which
was 15 days before the initiation of the lesions. Based on the history
of contact with the sheep, the appearance of the lesions, and the
physical examination, we clinically diagnosed the patient with orf
disease. Nose and finger skin biopsy performed for real-time
polymerase-chain reaction. Parapoxvirus infection was confirmed
by real-time polymerase-chain reaction at Refik Saydam National
Reference Laboratory. No specific treatment was initiated. Eight
weeks after the initiation, his lesions recovery completely without
any complications. The patient showed no signs of recurrence at the
end of a 1 year follow-up period.
DISCUSSION
Orf, also known as ecthyma contaginosum, scabby mouth, sore
mouth, or contagious pustular dermatitis is a zoonotic infection
caused by parapoxvirus that commonly infects humans with contact
infected animals. Orf virus usually causes self-resolving cutaneous
lesions. Progressive lesions can develop in immunocompromised
patients.1
Hands and fingers are the most common sides of orf skin lesions,
with other sites such as the nose only occasionally being involved.2
Orf disease has been reported worldwide. Shepherds, sheep-
shearers, farmers, butchers, cooks, and veterinarians are at an
From the Department of Otorhinolaryngology, Gaziantep 25 Aralik State
Hospital, Gaziantep; and yNational Tularemia Reference Laboratory,
Public Health Institution of Turkey, Ankara, Turkey.
Received September 20, 2016.
Accepted for publication November 2, 2016.
Address correspondence and reprint requests to Nurdoğan Ata, MD, Depart-
ment of Otorhinolaryngology, Gaziantep 25 Aralik State Hospital,
Sarigüllük Mah. Fevziçakmak bulvari, PK:27060 Şehitkamil, Gaziantep,
Turkey; E-mail: nurdoganata@mynet.com
The authors report no conflicts of interest.
Copyright # 2017 by Mutaz B. Habal, MD
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000003441
# 2017 Mutaz B. Habal, MD
Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Brief Clinical Studies
FIGURE 1. (A) A 2-cm nodular mass with erythema in the nasal tip at day 15 of
infection. (B) Papule lesion on the digit of right hand.
increased risk for orf virus infection. An epidemic outbreak of orf
disease may be observed after the feast of sacrifice.3 In our patient
transmission from sheep was observed on the first day of the feast.
Treatment options of lesions have not been well characterized
duo to spontaneous recover. Usually, treatment may not be necess-
ary duo to the self-resolving. Some patients may become infected
with bacteria and some patients may develop progressive. Treat-
ment includes surgical excision, antiviral drugs, and topical anti-
biotic therapy. Imiquimod, cidofovir, and intralesionel interferon
alpha can be used in immunocompromised patients.1,4 In the present
patient, treatment may not be necessary, given the spontaneously
recovery.
In conclusion, orf is transmitted by direct contact with infected
animals. In addition to the farmers, veterinarians, and butchers,
those who contact sheep at the feast of the sacrifice in the muslim
countries may be affected. Persons who contact with sheep at the
feast of the sacrifice should use gloves and avoid exposure of open
wounds. Although orf lesions mostly occur in the hands, nose orf is
extremely rare. In a patient with nose skin lesion, nose orf must be
kept in the mind that as a differential diagnosis in patients who have
a history of contact with animals.
REFERENCES
1. Lederman ER, Green GM, DeGroot HE, et al. Progressive ORF virus
infection in a patient with lymphoma: successful treatment using
imiquimod. Clin Infect Dis 2007;44:e100–e103
2. Gurel MS, Ozardali I, Bitiren M, et al. Giant orf on the nose. Eur J
Dermatol 2002;12:183–185
3. Veraldi S, Nazzaro G, Vaira F, et al. Presentation of orf (ecthyma
contagiosum) after sheep slaughtering for religious feasts. Infection
2014;42:767–769
4. Al-Qattan MM. Orf infection of the hand. J Hand Surg Am
2011;36:1855–1858
The Study of Calcium,
Phosphonium, Magnesium, and
Ferrum Concentration in Serum
of Patients With Primary
Trigeminal Neuralgia
Hua Zhao, MD, Yinda Tang, MD, Xin Zhang, MD,
and Shiting Li, MD
Objective: To measure the concentration of 4 essential elements
(Ca, P, Mg, Fe) in serum of patients with primary trigeminal
e235
Brief Clinical Studies The Journal of Craniofacial Surgery
neuralgia. And evaluate the role of the 4 elements in serum on the
pathogenesis of trigeminal neuralgia (TN), and the relevance of
etiology.
Methods: From June 2013 to June 2014, a number of 80 patients
with primary trigeminal neuralgia were collected. We present a
retrospective review of the concentration of 4 essential elements
with those of control group of 80 patients without TN. The
concentrations of 4 elements were measured with Olympus AU
400 automatic biochemistry analyzer.
Results: In primary TN patients, the concentration of Ca, P, Mg
in serum is lower than the control group obviously (P <0.05),
but the concentration of Fe in serum is higher than the control
group obviously (P <0.05). Along with level of the VOS
improved, the concentration of Ca, P, and Mg in serum becomes
lower and lower. However, along with level of the VOS
improved, the concentration of Fe in serum becomes higher
and higher.
Conclusion: Patients with primary TN, the change of 4 elements
concentration in serum is connected with biochemical material
change. The change of the microelement may play an important
role in the development of Trigeminal neuralgia, possibly demye-
linating lesions.
Key Words: Calcium, ferrum, magnesium, phosphonium, primary
trigeminal neuralgia
P rimary trigeminal neuralgia (PTN) is a chronic pain syndrome
characterized by recurrent, paroxysmal shock-like, stabbing in
the distribution area of 1 or more branches of trigeminal nerve. The
incidence of PTN is about 4 to 5 per 100,000.1– 3 It is the most
common reason for facial pain in middle-aged and elderly people.
However, the reason of PTN is unclear. Dandy4 first covered the
relationship of vascular compression with primary trigeminal neur-
algia. Jannetta5 developed and popularized the theory of the vas-
cular compression. But, the absence of culprit vascular in PTN has
also been covered.6,7 So far the related research mainly focused on
choices of modus operandi and the reports of microelement in PTN
were rare.
In the present study, we measured the concentration of 4
essential elements (Ca, P, Fe, and Mg) in serum of patients with
primary trigeminal neuralgia. And evaluate the role of the 4
elements in serum on the pathogenesis of TN, and the relevance
of etiology.
From the Department of Neurosurgery, Xinhua Hospital, Shanghai Jiao
Tong University School of Medicine, Shanghai, China.
Received October 8, 2016.
Accepted for publication November 2, 2016.
Address correspondence and reprint requests to Shiting Li, MD,
Department of Neurosurgery, Xinhua Hospital, Shanghai Jiao Tong
University School of Medicine, 1665 Kongjiang Road, Yangpu District,
Shanghai 200092, China; E-mail: xinhuaneuro@126.com
HZ and YT are co-first authors.
This study was supported by Shanghai Xinhua Hospital Foundation
(15LC21), Shanghai Municipal Health Bureau (2013SY019), and
Science and Technology Commission of Shanghai Municipality
(14D21930303).
The authors report no conflicts of interest.
Copyright # 2017 by Mutaz B. Habal, MD
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000003443
e236
Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
 Volume 28, Number 3, May 2017
METHODS
Patients and Population
From June 2013 to June 2014, a number of 80 patients (group A)
with primary trigeminal neuralgia were collected in the Department
of Neurosurgery at Xing Hue Hospital, Shanghai Jiao Tong Uni-
versity School of Medicine, Shanghai, China. At same time, a
number of 80 patients (group B) without primary trigeminal neur-
algia were collected. The group B was defied as control group. Main
inclusive criteria: all patients were treated for the first time; normal
hepatic function; without taking medication that affects the metab-
olism of calcium, phosphorus, ferrum, magnesium in last 2 months;
without closely contact history with calcium, phosphorus, ferrum,
magnesium in work environment; without disease that affect the
metabolism of calcium, phosphorus, ferrum, magnesium.
This study and the technique were approved by Xin Hua
Hospital Ethics Institutional Committee.
Methods
The pain sensation was evaluated with visual analogous scale
pain score. I level: no pain; II level: slight pain; III level: severe
pain; IV level: worst possible.
Blood-electrolytes, renal functions, hepatic function, were mon-
itored at hospitalization with fast blood each patient. The concen-
trations of four essential elements were measured with Olympus
AU400 automatic biochemistry analyzer.
Statistical Analysis
SPSS 18.0 was used for statistical analysis; all the results were
described as mean value  standard deviation. P  0.05 indicated
statistical significance. Student t test and x2 test were performed
to compare the mean continuous data and the categorical data
respectively.
RESULTS
In group A: the average age of patients was 61.23  2.05 years
(range: 42–78); 50 patients were man and 30 patients were female.
In the group B: the average age of patients was 60.26  3.42 years
(range: 39–75); 49 patients were man and 31 patients were female.
The sex and age of 2 groups were no difference on statistics
(P > 0.05).
In group A: 15 patients were in level II, 28 patients were in level
III, 37 patients were in level IV. The concentrations of Ca, P, Mg in
serum were presented in Figure 1. The more serious pain, the lower
concerntration of Ca, P, Mg in serum. The concentration of Fe was
displayed in Figure 2. The more serious pain, the higher concern-
tration of Fe in serum. There are significant differences between the
there difference levels (P < 0.05).
FIGURE 1. The concentration of Ca, P, Mg in serum. The figure presents that the
more serious pain, the lower concerntration of Ca, P, Mg in serum.
# 2017 Mutaz B. Habal, MD
The Journal of Craniofacial Surgery  Volume 28, Number 3, May 2017
FIGURE 2. The concentration of Fe in patients with TN. The figure presents that
the more serious pain, the higher concerntration of Fe in serum.
In group A: the concentrations of Ca, P, Mg, Fein serum
were 2.35  0.08, 0.99  0.03, 0.88  0.05, 15.03  1.89 mmol/L
respectively. In group B: the concentrations of Ca, P, Mg, and Fe in
serum were 2.45  0.11, 1.21  0.09, 0.97  0.08, and 13.78  0.83
mmol/L respectively.
There were significant differences in 2 groups on the concen-
tration of Ca, P, Mg, and Fe in serum (Table 1).
DISCUSSIONS
Primary trigeminal neuralgia is a common clinical disease. The
characteristic manifestation is a serious bursting pain of the face. In
PTN, pain episodes that may happen at any time usually continue
from seconds to minutes. In additional, the pain episodes are not
only triggered by motions (brushing or chewing) but also sensorial
stimulation of faces.8,9
There are many treatments of trigeminal neuralgia, but the
results of the treatments were barely satisfactory. In other words,
although many treatments, but no one recognized ideal means, the
difficulty to treat the disease is that the pathogenesis of trigeminal
neuralgia is not clear.10,11 There are many classical theories of PTN,
including peripheral hypothesis, central hypothesis.5 Autoimmune
theory and biochemical doctrine are all the immerging theories.
However, most scholars considered that PTN is a multifactor
disease. The changes of demyelization of peripheral nerve, with
the participation of immune factor, are the pathological basis of
PTN. The weakened inhibitory action of spinal nucleus of trigem-
inal nerve is an important condition for PTN. Most of the research in
the past focused on the choices of modus operandi for PTN;
however, the new research method, research aspects of basic
research about PTN are sorely missing.
With the development of detecting technique and research
methods, more and more research contents focused on various
trace elements. Trace elements associate with a number of
diseases. Cuajungco and Goldstein12 reported that the lack of
zinc can lead to Alzheimer disease. Parkkila and Parkkila13
reported that the level of Carbonic anhydrate II was moderately
increased in patients with central nervous system infection,
dementia, or PTN. But there is a little study that trace elements
associate with PTN.
TABLE 1. The Concentration of Ca, P, Mg, and Fe in Serum
Ca (mmol/L) P (mmol/L) Mg (mmol/L) Fe (mmol/L)
Group A 2.35  0.08 0.99  0.03 0.88  0.05 15.03  1.89
Group B 2.45  0.11 1.21  0.09 0.97  0.08 13.78  0.83
P value <0.05 <0.05 <0.05 <0.05
# 2017 Mutaz B. Habal, MD
Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Brief Clinical Studies
Relationship Between Ca, P, and Primary
Trigeminal Neuralgia
Calcium is a necessary nutrient element for humans, as it helps
to bone mineralization and rigidity, thus preventing problems such
as osteoporosis and fractures in adulthood and old age.14 And it also
has an important role in fibrin formation during the blood coagu-
lation process and regulation of muscle contraction. In addition, it
has distinct characteristics that allow it to explicitly activate differ-
ent cellular responses.15 The physical activities such as neuromus-
cular excitement, nerve impulse conduction, and normal heart
beating are related closely to ionized calcium. Phosphorus is second
only to calcium in its abundance in humans. Phosphorus and
calcium are absorbed in the jejunum, and deposited in bone. There
is a very close relationship between phosphorus and calcium. The
calcium and phosphorus metabolism are affected by calcitonin. The
result of CaP is an invariant constant.
In the present study, we discovery that the content of Ca and P in
TN is lower than those in control group. It proved that the patients
with TN have bone abnormal metabolism. Multiple factors
regulated the bone metabolism, and calcitonin plays the most
important role in bone metabolism. Goadsby reported that TN is
closely related to trigeminovascular system (TVS). The TVS is
activated in TN and it can result in the decline of Ca and P in
serum.16 Our results are in accordance with Goadsby.
Relationships Between Mg, Fe, and Primary
Trigeminal Neuralgia
Mg is one of the essential trace elements that play important
roles in normal physiology of human body. It is participative in
many important physiological activities such as cell proliferation,
differentiation, nucleic acids, and protein metabolism. It breaks
down cholesterol and fats in the blood stream, thus helping to
maintain healthy normal cholesterol levels. Fe is also one of the
essential trace elements. It plays a vital role in maintaining structure
and function of lymphocytes. It can enhance phagocytosis and
bactericidal activity of neutrophils. Drueke et al17 reported that
Fe is involved in atherogenesis, thrombosis, and apoptosis. In the
present study, we get a result that the content of Mg in TN was
markedly lowered and the content of Fe in TN was markedly
increased, which can lead to hypertension. Hypertension can cause
the endothelial dysfunction that can result in atherogenesis. Hilton
and Love18 have covered that atherogenesis can cause trigeminal
demyelination. So we think that the one of the reasons of trigeminal
demyelination is the declined content of Mg and the raised content
of Fe.
CONCLUSIONS
The content of Ca, P, and Mg in patients with PTN was obviously
reduced, and the content of Fe in patients with PTN was signifi-
cantly increased. The change of microelement may play an import-
ant role in the development of PTN, possibly demyelinating
lesions.
REFERENCES
1. Zakrzewska JM, Coakham HB. Microvascular decompression for
trigeminal neuralgia: update. Curr Opin Neurol 2012;25:296–301
2. Larsen A, Piepgras D, Chyatte D, et al. Trigeminal neuralgia: diagnosis
and medical and surgical management. JAAPA 2011;24:20–25
3. Oomens T. Forouzanfar: pharmaceutical management of trigeminal
neuralgia in the elderly. Drugs Aging 2015;32:717–726
4. Dandy WE. Concerning the cause of trigeminal neuralgia. Am J Surg
1934;24:447–455
5. Jannetta PJ. Arterial compression of the trigeminal nerve at the pons in
patients with trigeminal neuralgia. J Neurosurg 1967;(suppl):159–162
e237
Brief Clinical Studies The Journal of Craniofacial Surgery
6. Revuelta-Gutie rrez R, Lopez-Gonzales MA, Soto-Hernandez JL.
Surgical treatment of trigeminal neuralgia without vascular
compression: 20 ears of experience. Surg Neurol 2006;66:32–36
7. Baechli H, Gratzl O. Microvascular decompression in trigeminal
neuralgia with no vascular compression. Eur Surg Res 2007;39:
51–57
8. Oesman C, Mooij JJ. Long-term follow-up of microvascular
decompression for trigeminal neuralgia. Skull Base 2011;21:
313–322
9. Jia DZ, Li G. Bioresonance hypothesis: a new mechanism on the
pathogenesis of trigeminal neuralgia. Med Hypotheses 2010;74:505–507
10. Tucer B, Ekici MA, Demirel S, et al. Microvascular decompression
for primary trigeminal neuralgia: short-term follow-up results
and prognostic factors. J Korean Neurosurg Soc 2012;52:42–47
11. Li ST, Pan Q, Liu N, et al. Trigeminal neuralgia: what are the important
factors for good operative outcomes with microvascular
decompression? Surg Neurol 2004;62:400–404
12. Cuajungco MP, Goldstein LE. Evidence that the beta-amyloid plaques
of Alzheimer’s disease represent the redox-silencing and entombment of
abet by zinc. J Biol Chem 2000;275:19439–19442
13. Parkkila AK, Parkkila S. Carbonic anhydrate II in the cerebrospinal
fluid: its value as a disease marker. Eur J Clin Invest 1997;27:
392–397
14. Allodi I, Udian E. Specificity of peripheral nerve regeneration:
interactions at the axon level. Progress in neurobiology 2012;98:
16–37
15. Courjaret R, Machaca K. Mid-range Ca2t signalling mediated by
functional coupling between store-operated Ca2t entry and IP3-
dependent Ca2t release. Nat Commun 2014;5:3916.
16. Goadsby PJ, Edvisson L. Human in vivo evidence for trigeminovascular
activation in cluster headache. Neuropeptide changes and effects of
acute attacks therapies. Brain 1994;117:427–434
17. Drueke T, Witko-Sarast V. Massy: iron therapy, advanced oxidation
protein products and carotid artery intima-media thickness in end-stage
renal disease. Circulation 2002;106:2212–2217
18. Hilton DA, Love S. Pathological finding associated with trigeminal
neuralgia caused by vascular compression. Neurosurgery 1994;35:
299–303
Rehabilitation of the Work
Accident-Related Traumatic
Mandible With Iliac Free Flap,
Distraction Osteogenesis, and
Dental Implants
Yavuz Findik, DDS, PhD, Timuçin Baykul, DDS, PhD,
Mustafa Asim Aydin, DDS, PhD,y Selman Altuntaş, DDS, PhD,y
and Zeynep Başağaog˘lu Demirekin, DDS, PhDz
Abstract: Mandibular bone defects due to extensive trauma impair
occlusion and affect the aesthetics of facial contouring, making it
difficult to obtain a satisfactory outcome. Distraction osteogenesis
and free flap is an effective and aesthetic treatment option for
rehabilating these defects. In this clinical report, the authors present
rehabilitation of a wide mandibular traumatic defect due to a work-
related accident with iliac free flap, distraction osteogenesis, and
dental implants.
Key Words: Distraction osteogenesis, free flap, mandible
e238 # 2017 Mutaz B. Habal, MD
Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
 Volume 28, Number 3, May 2017
T raumatic mandibular injuries were frequently seen in many
medical centers.1 Simple mandibular fractures were generally
treated with open or closed reduction methods, conversely wide
traumatic mandibular defects were treated with more complicated
techniques such as free flaps or distraction techniques. The main
limitation of the free flaps for reconstruction of the mandible is the
insufficient bone height for the reconstruction of both the skeletal
base and the alveolar ridge.2 Hence, vertical distraction osteogen-
esis is a good choice for achieving both aesthetic and functional
outcomes after the free flap healing. In this clinical report, we
present rehabilitation of a wide mandibular traumatic defect due to a
work-related accident with iliac free flap, distraction osteogenesis,
and dental implants.
CLINICAL REPORT
A 32-year-old male patient was referred to our center with a wide
mandibular defect due to a work-related accident. After clinical and
radiographical examination, wide defect with multiple fractures due
to high-energy trauma was seen on the right side of the mandible
(Fig. 1A). First, a reconstruction plate was applied with other plates
and wires for treating the comminuted mandible fracture (Fig. 1B).
For better both aesthetic and functional results, reconstruction plate
was removed and free iliac flap was applied to the wide defect area.
One year after the reconstruction, to gain sufficient bone height for
dental implants, vertical distraction osteogenesis of the iliac and
mandibular flap was performed (Fig. 1C). Under general anesthesia,
intraoral incision was made on the right mandibular region. The
labial and buccal periostea were reflected, but the lingual perios-
teum and soft tissue were not elevated. A wide osteotomy was
performed right molar area to the left canine region, and an
extraosseous large vertical distraction device (KLS Martin, Jack-
sonville, FL) was placed on the vestibular surface of the mandible.
The device was fixed with unicortical microscrews and was
temporarily activated to ensure correct function (Fig. 1D). An
antibiotic was administered intravenously for 2 days and continued
orally for 5 days. The device was activated at a rate of 0.5 mm/day
from the seventh postoperative day. Four implants (OsseoSpeed;
Astra Tech AB, Molndal, Sweden) were placed 4 months after
distraction (Fig. 2A). The implants were loaded 3 months after
implantation, and an implant-supported fixed denture was fabricated.
The final prosthesis was followed up to 1 year, with stable conditions
(Fig. 2B).
DISCUSSION
Comminuted mandibular fractures have been treated by several
methods: closed or open reduction methods. For wide comminuted
mandibular fractures, other techniques such as distraction osteo-
genesis or free flaps were used for rehabilating the large traumatic
gaps.3
Iliac free flap is effective in providing both good functional and
aesthetic results for reconstruction of mandibular bone defects. The
From the Department of Oral and Maxillofacial Surgery; yDepartment of
Plastic and Reconstructive Surgery; and zDepartment of Prosthodontics,
Faculty of Dentistry, Süleyman Demirel University, Isparta, Turkey.
Received October 11, 2016.
Accepted for publication November 2, 2016.
Address correspondence and reprint requests to Dr Yavuz Findik, DDS,
PhD, Department of Oral and Maxillofacial Surgery, Faculty of Den-
tistry, Süleyman Demirel University, Isparta, Turkey;
E-mail: yavuzfindik32@hotmail.com
The authors report no conflicts of interest.
Copyright # 2017 by Mutaz B. Habal, MD
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000003466