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PubMed2005

Lymphatic malformation of the lingual base and oral floor.

BACKGROUND: Lymphatic malformation of the tongue and floor of the mouth is associated with chronic airway problems, recurrent infection, and functional issues related to speech, oral hygiene, and malocclusion. There are no accepted anatomic guidelines or treatment protocols. METHODS: This retrospective review focused on anatomic extent, treatment, complications, and airway management in 31 patients with lymphatic malformation of the lingual base and oral floor. RESULTS: Involved adjacent structures included the neck (77 percent), mandible (41 percent), face (42 percent), lips (10 percent), pharynx (45 percent), and larynx (26 percent). Fifty-eight percent of patients required tracheostomy during infancy; decannulation was possible in two-thirds of these patients. Management included resection alone (42 percent), resection and sclerotherapy (26 percent), resection and laser coagulation (16 percent), sclerotherapy and laser coagulation (16 percent), and resection and radiofrequency ablation (3 percent). Resection involved the neck (58 percent), floor of the mouth (52 percent), and tongue (42 percent); there were often multiple procedures. Aspiration was tried with little success in 10 percent of patients. Virtually all patients had residual abnormal lymphatic tissue. Complications and posttherapeutic problems included infection (81 percent), neural damage (27 percent), difficulty in speech (23 percent), feeding problems (10 percent), and seroma or hematoma (6 percent). Associated dental/orthognathic conditions, particularly prognathism and anterior open bite, were documented in one-third of patients. CONCLUSIONS: The initial step in the protocol is control of the neonatal airway. Staged cervical resection is undertaken in late infancy to early childhood; resection should also include abnormal tissue in the oral floor. Sclerotherapy is primarily for macrocystic disease or secondarily for recurrent cysts following partial extirpation. Vesicles of the mucous membranes and dorsal tongue are treated either by sclerotherapy, laser (carbon dioxide, yttrium-aluminum-garnet, or potassium-titanyl-phosphate), or radiofrequency ablation. Reduction for macroglossia is indicated for persistent protrusion or to allow correction of malocclusion. Embolization controls lingual bleeding. Orthognathic procedures are undertaken at the appropriate age, only after lingual size and position are acceptable.

Catheter Ablation↗
PubMed1976

[The aglossia-adactylia syndrome].

We present two own cases of the aglossy-adactyly syndrome and a review of 25 cases of the literature. Our experience concernes two girls. One was followed from birth until 4 1/2 years, the other from 5 to 9 years. This helped us to obtain a good knowledge of the development of these patients. Malformations of the mouth and of the limbs with a normal intelligence characterize the aglossy-adactyly syndrome. The most important features are: shortness of the tongue of variable degree, anomaly of the teeth, hypoplasia of the mandible and ectromelia of different extension with a peripheric predilection and often with an asymmetry. The etiology is unknown. All the known cases are sporadic. The differential diagnosis of the aglossy-adactyly syndrome includes the ankyloglossia superior, the Hanhart syndrome and the oro-farcio-digital syndrome I. Even when the mouth malformations are of a great degree, the functional limitations of the patients with aglossy-adactyly syndrome are very small. This is important in determining the prognosis, in consulting the patients and their parents and for decisions about corrective operations.

Abnormalities, Multiple↗
PubMed2005

Tolerance of deviations in eye and mouth position.

OBJECTIVE: To develop and refine a technique for measuring the tolerance for deviations in facial appearance. DESIGN: A psychophysical method was administered using photocopies of altered facial photographs to measure tolerances for deviations in eye position and mouth angulation and judges' reaction time. METHODS: Stimulus photos were displayed as Kodachrome slides to 76 individuals grouped by their familiarity with craniofacial anomalies, i.e. dental professionals, orthodontic and craniofacial patients, also a group of normal patients. RESULTS: (1) Tolerance for deviation of facial appearance varies inversely with the magnitude of the physical deviations from normal; (2) tolerance varies directly as a function of assumed familiarity with deviation; (3) response and reaction time varied inversely with the tolerance for facial deviation. CONCLUSION: Significant differences in tolerance and reaction time were found among the groups and depended on whether an isolated feature was judged or if the feature was judged in the context of the whole face.

Adolescent↗
PubMedDate not supplied

Oromandibular limb hypogenesis syndrome.

A few cases have been presented regarding Hypoglassia Congenita with anterior maxillo-mandibular fusion. Our case is reported as Hypoglassia Congenita but with complete maxillo-mandibular fusion associated with hypodactylia. This condition presented as a problem of not being able to open the mouth, so there is no capacity for feeding and, more so, speech.

Abnormalities, Multiple↗
PubMed1993

D8S7 is consistently deleted in inverted duplications of the short arm of chromosome 8 (inv dup 8p).

Ten patients with inverted duplication of 8p (inv dup 8p) were studied with cytogenetic, biochemical and molecular techniques. The duplication for the region 8p12-p22 was always associated with a deletion of the locus D8S7 (mapped in 8p23.1) as demonstrated with the probe pSW50 by both in situ hybridization and Southern blot. Restriction fragment length polymorphisms detected by probes pSW50 (1 case) and by pG2LPL35 (locus LPL) (two cases) were informative as to a maternal origin of the anomaly. The activity of glutathione reductase, whose gene maps in the duplicated region at 8p21.1, was increased in all patients. The recognizable phenotype of inv dup 8p includes neonatal hypotonia, prominent forehead, large mouth with everted lower lip, abnormally shaped large ears, brain malformations and severe mental retardation. Our findings indicate that the chromosome rearrangement is homogeneous at least for the presence of the deletion and support the hypothesis of a common mechanism of origin.

Abnormalities, Multiple↗
PubMed1993

Two Class II, division 1 patients with congenitally missing lower central incisors.

Although orthodontic treatment objectives and procedures for apparent protrusion of the maxillary teeth vary among orthodontists and specific cases, the differences are even greater where there is disharmony of jaw relationship between the maxilla and the mandible. The two cases presented in this article resemble each other in appearance, but the growth patterns and reactions to appliance, as well as treatment progress, are quite different. The initial excessive overjet seemed equally severe. Treatment was started at the same age, both patients were eldest daughters, both had a convex type facial pattern and an abnormal perioral muscle function with mouth breathing. In addition to the marked overjet, deep bite, and Class II molar relationship, lower central incisors were missing, and second molars had not erupted. They both had a steep curve of Spee, disharmony between the upper and lower arch forms, and slight diastemata. The facial appearance has been improved significantly in both, and patient satisfaction is high. A Kloehn-type cervical headgear was used in both cases. Case 1 showed improvement in the relationship between the maxilla and the mandible with reduction of the overjet. However, in Case 2, the sagittal relationship became worse, and an open bite developed. The obvious question is why the big difference in treatment response? What role did clearing of the nasopharyngeal airway play?

Activator Appliances↗
PubMed1997

Fluoxetine vs. tricyclic antidepressants in women with major depressive disorder.

Major depressive disorder and dysthymia are twice as prevalent in women as in men, and the lifetime risk of a woman's developing a major depressive disorder is about 20%. Yet depression is often unrecognized or misdiagnosed in women, and only about one quarter of women who meet criteria for major depressive disorder receive appropriate therapy. Until recently, women were generally excluded from clinical drug trials because of concerns of inadvertent pregnancy and risk of teratogenicity. Thus, information on safety and efficacy in those most likely to require antidepressant therapy is lacking. Studies have shown that the antidepressant fluoxetine, a selective serotonin reuptake inhibitor (SSRI) has a more tolerable side effect profile than do tricyclic amine (TCA) antidepressants, but few data have been reported on the efficacy and tolerability of fluoxetine or other SSRIs in female patients. In this study, a retrospective analysis of 11 randomized, double-blind, well-controlled trials was done to compare data from 427 female patients on fluoxetine and 423 female patients on TCAs. Both fluoxetine and TCAs significantly reduced the HAMD17 total mean score from baseline to end point, week 5 (fluoxetine, 24.35 to 14.37; TCAs, 24.57 to 14.43; p < 0.001). Both treatment groups were associated with significant reductions in the HAMD17 anxiety/somatization and insomnia subfactor scores. Abnormal vision, constipation, dizziness, dry mouth, and somnolence occurred more frequently (p < 0.05) in the TCA group. Insomnia and nausea were the only adverse events more common (p < 0.05) in the fluoxetine group. This study demonstrates that fluoxetine is an effective and tolerable agent for the treatment of major depressive disorder in women.

Adult↗
PubMed1995

Congenital shortening of the anterior lamella of all eyelids: the so-called ablepharon macrostomia syndrome.

The so-called ablepharon macrostomia syndrome is an extremely rate congenital condition that includes abnormal ears, an enlarged, fishlike mouth, absence of lanugo, redundant skin, and vertical shortening of all eyelids. Only four cases have been described so far. In these cases the nature of the eyelid anomalies has not been clearly defined. We report one more case showing that the condition is better described as a severe microblepharon because only the anterior lamella of the eyelids is shortened. The literature about this condition is reviewed, and oculoplastic treatment is discussed.

Adolescent↗
PubMed1978

Delayed small-intestinal transit in tropical malabsorption.

The time of breath hydrogen appearance (T) after oral lactulose was determined in 98 patients in London who had been to the tropics. Fifty-six controls from three different ethnic groups had no evidence of gastrointestinal disease; 23 had tropical malabsorption, which was severe in 10; and 19 had chronic diarrhoea without malabsorption. Mean T was significantly delayed in patients with tropical malabsorption compared with controls, indicating that their mouth-caecum transit rate was abnormally slow. In six patients with tropical malabsorption who were reinvestigated after treatment a fall in the value for T seemed to parallel clinical improvement. Three patients with diarrhoea due to chronic colonic disease had delayed T. Patients with tropical malabsorption have bacterial colonisation of the small intestine, which is important in the pathogenesis of the disease. Bacterial overgrowth, which sometimes occurs after infective diarrhoea in the tropics and gives rise to tropical sprue, is a result of stasis.

Administration, Oral↗
PubMed1994

Adjuvant breast disease: an evaluation of 100 symptomatic women with breast implants or silicone fluid injections.

We evaluated 100 referred women with breast implants (n = 97) or silicone fluid injections (n = 3) into breasts who developed various symptoms. All reported symptoms occurred at a median latency period of 6 years (range 0-24 years) after implantation or injection of silicone. Commonest symptoms were weakness (95%), fatigability (95%), myalgia (90%), morning stiffness (89%), arthralgia (81%), memory loss (81%), sensory loss (77%), headache (73%) and dry eyes and dry mouth (72%). Laboratory results revealed abnormal levels of serum immunoglobulins or complement in 57% and autoantibodies in 78%. Sural nerve biopsy was abnormal in 80% with the major finding of loss of myelinated fibers in 79%. Biceps muscle biopsy was abnormal in 58% with the major finding of neurogenic atrophy in 27%. Ninety-six patients underwent implant removal; 60% of the patients were found to have one or both implants ruptured with silicone spilled into tissue. At time of removal, a pectoralis major muscle biopsy was taken which was abnormal in 89% with the major finding of neurogenic atrophy in 55%. Biopsy of implant capsule was abnormal in 94% showing foreign body giant cells containing refractile material consistent with silicone in 69% whether or not the elastomer shell was ruptured. Silicone can cause a systemic autoimmune disease with a variety of symptoms probably due to a global activation of the immune system. Since our patients had objective laboratory and histologic findings together with a high rate of mechanical implant failure, further investigations are necessary.

Adult↗
PubMed1988

[A case report of nasal infestation by the leech, Dinobdella ferox].

A 55-year-old man in Okagaki Town, Fukuoka Prefecture, pulled a nasal leech from his nostril in July 1987, after suffering from nosebleed, copious running snivels as well as unpleasant foreign body sensation in the nasal cavity. Except for nasal septum deviation, no abnormality in the ears and mouth cavity nor bleeding, ulcerous and erosive changes in the nasal cavity were found. The formalin-fixed specimen of the leech was nearly black with no definite stripes or spots, and measured 3.5 cm in length and 1.2 cm in width. Because of these and the following characteristics, viz. 1) auricles on the posterior segment were absent, 2) five pairs of eyes were present in the anterior segments with the eye pairs 3 and 4 separated by an annulus, and 3) teeth were not observed, this specimen was identified as Dinobdella ferox. This nasal leech is found widely in Southeast Asia. In Japan, some human cases of its infestation have also been reported, mainly from southern Kyushu. The leech seemed to have entered the nasal passage of the present case from a stream at a hot spring in northern Kyushu. Attention should be given to nasal leech infestation especially now that many people in Japan are eager to visit isolated hot spring resorts.

Animals↗
PubMedDate not supplied

Orthodontic diagnosis in young children: beyond dental malocclusions.

Orthodontics is more than merely diagnosing and treating dental malocclusion. It also includes diagnosis and treatment of facial/skeletal problems, upper airway obstruction (mouth breathing), temporomandibular dysfunction, and abnormal myofunctional habits. These assessments should be made in all patients but especially in children as young as age 5 to take advantage of facial growth. If problems are diagnosed early and preventive measures are instituted, many facial and dental problems may be minimized or averted.

Airway Obstruction↗
PubMed1991

Ultrasonography of the salivary glands in the evaluation of Sjögren's syndrome. Comparison with sialography.

Ultrasonography (US) was evaluated as noninvasive method to determine salivary gland involvement and compared with sialography in patients with Sjögren's syndrome (SS) and patients with only objective xerostomia and/or xerophthalmia. Patients with complaints of dry eyes and dry mouth but with no objective abnormalities served as control group. Ultrasound revealed a decreased parotid gland reflectivity in most of the patients with SS, compared with only 2 patients in the other group (p = 0.002). Our study suggests that US can be a useful, noninvasive method in the diagnostic work-up of patients with sicca complaints.

Humans↗
PubMed1988

Ultrasonography and computer tomography of the salivary glands in the evaluation of Sjögren's syndrome. Comparison with parotid sialography.

Ultrasonography (US) and computed tomography (CT) were studied as noninvasive methods to determine salivary gland involvement and compared with parotid sialography in patients with Sjögren's syndrome (SS) and patients with only objective xerostomia and/or xerophthalmia. Patients with complaints of dry eyes and dry mouth but with no objective abnormalities served as controls. CT was not discriminative between the 2 groups. In contrast, US revealed decreased parotid gland echogenicity in most of the patients with SS, compared with only 1 patient in the group with sicca complaints only (p = 0.005). Our study suggests that US can be a useful, noninvasive method in the diagnostic investigation of patients with sicca complaints.

Humans↗
PubMed1994

Megalocornea, developmental retardation and dysmorphic features: two further patients.

Two unrelated children are described with megalocornea, mild-moderate developmental delay, mild joint laxity and a similar dysmorphic appearance, comprising a bossed forehead, hypertelorism, a saddle-shaped nose and a carp-shaped mouth with prominent lips. Similar abnormalities have been observed in previously reported cases of megalocornea/mental retardation and may help to define one subtype of this heterogeneous group of conditions.

Abnormalities, Multiple↗